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We report a 47-year-old female patient with multiple intradural extramedullary spinal arachnoid cysts. The patient's symptoms started 3 years prior to the surgery with right dorsal radicular pain (D5–D6). She was admitted in a neurology department and was treated for herpes zoster with poor improvement of the pain. Two years after the first clinical symptoms, the patient presented with a progressive weakness of the left lower limb. The patient underwent a whole spine MRI where dorsal MRI showed multiple intradural arachnoid cysts which caused an important spinal cord compression [Figure and ]. She was followed conservatively in another hospital in a neurosurgical ward. She came to our center 3 years after the appearance of the first symptoms for progression of the neurological deficit in the left lower limb (walking became more difficult) and for the increase in radicular pain at D5–D6 on the right side. We explained to the patient the disease and the treatment options, that is, a wait-and-see approach or a surgical procedure with a total or a partial cyst removal. After careful evaluation of the discussed treatment options, the patient chose the surgical approach. The surgical procedure was uneventful and consisted of a bilateral laminoplasty of D5–D9 with a near total removal of the cysts, leaving only a part of the cyst wall that was tightly attached to the spinal cord. Intraoperatively, a diffuse thickening of the arachnoid and diffuse calcification were seen []. The aim of our procedure was to obtain a cranial-caudal liquoral communication, which was obtained at the end of the surgery when the spinal cord expanded []. The patient reported a complete resolution of symptoms in her left lower limb, however, in the first months an important radicular dorsal pain on the right side persisted. The radicular pain responded well to lidocaine plaster. In the radiological follow-up after 3 and 9 months after the surgery, the dorsal MRI showed a dilatation of the central canal with a localized intramedullary arachnoid cyst [Figure and ]. |
Herein we present a case of 62-year-old male who presented to our institution with a painful lump in his left breast with chronic sinus formation and pus discharge. According to the patient, the lump had been noticed for one month. The patient had loss of appetite and weight loss. The patient had a history of a common room with his colleagues during trip to an Asian country. The patient did not have any past or family history of pulmonary or extra-pulmonary tuberculosis. He was neither immune-compromised nor a smoker without any comorbidities. On physical examination, the left breast was tender, and an irregular mass of 3 x 2 cm was felt in upper outer quadrant of the left breast with multiple palpable axillary lymph nodes. Hematological and biochemical parameters were within normal limits, including negative testing for HIV. His initial workup included a mammogram which revealed irregular soft tissue density mass in upper outer quadrant of left breast (Figure ).\nThis was followed by ultrasound which showed irregular communicating branching sinus tracts (Figure ). These were found extending towards left axilla.\nThere were multiple enlarged lymph nodes in ipsilateral axilla with thickened cortices. The largest one was measuring 2.3 x 1.5 cm with cortical thickness of 0.98 cm (Figure ).\nA high possibility was raised for communication of these lymph nodes with the sinus tracts leading our diagnosis more towards tuberculosis.\nPatient's chest radiograph was normal without any evidence of parenchymal or pleural abnormality. The patient then underwent lymph node biopsy to rule out malignant etiology and to confirm the radiological diagnosis. Histopathology (images not available) revealed multiple epithelioid granulomas and multinucleated Langhan's type giant cells. Acute inflammatory exudate was also noted along with areas of caseous necrosis. There was no evidence of malignancy. It was concluded as chronic granulomatous inflammation with necrosis favoring tuberculosis. Histopathology was concordant with imaging and showed chronic granulomatous inflammation with necrosis. The patient received oral anti-tuberculosis therapy for six months without side effect or complications. Informed consent was taken from the patient before writing the case report. |
A 6-year-old female patient reported to the Department of Pedodontics and Preventive Dentistry, Government Dental College and Hospital, Hyderabad, with a complaint of swelling in the right side of the upper jaw. It was first noticed 6 months before the initial consultation. It was smaller in size and then suddenly increased to present size within 10 days. There was no history of fever, pain, sensory disturbance, bad taste or traumatic injury except discomfort and difficulty in mastication due to large swelling.\nExtraoral examination revealed an enormous swelling on the right side of the face involving the right maxilla causing obliteration of nasolabial fold and crossing the midline resulting in facial asymmetry (). The swelling had no localized elevation of temperature. There was no associated lymphadenopathy.\nIntraoral examination revealed diffuse swelling on the right side of maxilla obliterating the right buccal vestibule and crossing the midline extending up to deciduous lateral incisor of contralateral side. No teeth were visible intraorally on the effected side presumably due to pathological resorption caused by the lesion leading to loss of teeth.\nThe serial axial and coronal sections of computed tomography revealed lytic lesion causing destruction of cortex in right maxilla along with overlying large soft tissue component which was benign in nature ().\nLaboratory values for serum calcium, phosphorous, alkaline phosphatase and parathyroid hormone were within normal limits.\nIncisional biopsy obtained showed highly cellular tumor with nodular surface folds. The ulcer base revealed granulation tissue and dense chronic inflammatory infiltrate. The cellular lesion consisted of polygonal to spindle cells arranged in sheets and bundles admixed uniformly with many osteoclastic giant cells. The giant cells showed moderate amount of fibrillary pale vacuolated cytoplasm and oval to plump elongated vesicular nuclei. Mild to moderate nuclear pleomorphism and small nucleoli with increased mitotic activity in focal areas was suggestive of CGCG ().\nSurgical approach was preferred because of the size of the lesion (). Partial maxillectomy was performed via an intraoral approach under general anesthesia. As all the soft tissues involved in the lesion had to be removed it became imperative that soft tissue incisions were made through them down to the bone. To facilitate this, a sharp probe was used to determine and mark out the extent of the bony defect; incisions were subsequently made at least 1 cm away from the margins of the bony defect.\nAfter reflection of the soft tissue a fissure bur was used to cut the cortical bone around the lesion approximately 0.5 cm from its margin. The lesion was reflected in toto with the associated tissues and was removed completely (). Sutures were placed and patient was recalled after 2 weeks for suture removal (). There after she was recalled after every 1 month. The lesion healed completely after 6 months () following which reconstructive procedure was carried out uneventfully. |
A 32-year-old female was evaluated in our emergency department for the chief complaint of right-sided pulsatile tinnitus. The patient stated that her tinnitus had begun six months prior and was initially low in amplitude and only noticeable when using a stethoscope in her work as a registered nurse. However, her tinnitus became progressively louder over the two months before presentation, thus prompting her to seek medical evaluation. She noted several associated symptoms, including persistent headache, an irritating audible “whooshing” sound, and more recently a transient globus sensation. She denied any history of significant head or neck trauma. Her medical history was notable only for unchanged, chronic neck pain due to preexisting cervical spondylosis and she reported no known family history of hereditary diseases. She took low-dose aspirin daily and was undergoing hormonal therapy in preparation for in vitro fertilization, though she did not specify the medications involved in treatment.\nOn examination, a palpable thrill and loud bruit were evident over the right neck below the angle of the jaw. The patient's vital signs were within normal limits and she was neurologically intact. The ultrasound and computed tomography angiogram (CTA) of the neck demonstrated abnormal vertebral artery anatomy bilaterally, with a dilated and tortuous right vertebral artery and a smaller left vertebral artery entering the cervical foramen at C4. The CTA also demonstrated multiple dilated and serpiginous vessels surrounding the right vertebral artery at the level of C1 and opacification of the right internal jugular vein and epidural venous plexus on the right, suggesting a potential arteriovenous fistula (AVF) involving the right vertebral artery (Figures and ). The Department of Neurosurgery was consulted for further evaluation and treatment.\nThe patient was admitted to the neurosurgery service and diagnostic angiography the next day confirmed the presence of a fistula. On day two of admission, she underwent balloon test occlusion followed by endovascular sacrifice of the right vertebral artery and coil embolization of the fistulous communication (). She tolerated the procedure well and had immediate resolution of her pulsatile tinnitus, though she had some difficulty with postprocedural pain. On day five of admission, once established on an appropriate regimen of oral analgesics, she was discharged home in good health. At follow-up, the patient reported a right-sided headache originating at the occiput and radiating forward, but she remained neurologically intact, and angiography three months after operation confirmed successful obliteration of the fistula. |
A 60-year-old male was referred to our department, complaining about gradually worsening hoarseness, during the last 8 month period. Occasional dysphagia and foreign-body sensation were also reported upon referral. The patient was a heavy smoker for more than 20 years, reporting an average of 20 cigarettes per day. Alcohol was also a factor, and although no real alcohol abuse or indulgence was noted, the patient was a rather frequent user.\nMedical history only revealed arterial hypertension under treatment with beta blockers. Haematological and biochemical tests did not show any significant abnormalities.\nPhysical examination included a full head and neck examination, complemented with flexible fiberoptic laryngoscopy. Typical ear, nose and throat examination did not reveal any abnormal findings and neck palpation was negative. However, fiberoptic laryngoscopy revealed a lesion affecting both vocal cords and anterior commissure, while vocal cord mobility appeared impaired. On these grounds, a cervicothoracic and upper abdomen computed tomography (CT) scan with intravenous gadolinium was decided and the patient was scheduled for direct microlaryngoscopy and biopsy of the lesion under general anaesthesia.\nImaging confirmed the laryngeal lesion, yet it also indicated a second lesion about 2 cm below the inferior end of the primary one, arising somewhere between the first and second tracheal ring. Intermediate tissue appeared grossly normal (). No signs of enlarged cervical lymph nodes were noted and laryngeal cartilages showed no abnormal findings.\nOn the other hand, histopathological examination after biopsy of the lesion under general anaesthesia confirmed the diagnosis of squamous cell carcinoma. The lesion was carefully mapped and proved to be a glottic carcinoma affecting the anterior commissure and appearing in strong correlation with the thyroid cartilage. The lesion infiltrated the left and the first tertile of the right vocal cord. No subglottic extension was noted. In this context, the patient was informed and consent for radical surgical therapy was obtained.\nThe patient underwent total laryngectomy and wide excision of the trachea which included the second tumour within safe limits (). The procedure was complimented with left thyroid lobectomy and bilateral selective neck dissection (Robin’s levels II–IV). Paratracheal lymph nodes (Robin’s level VI) were also carefully dissected. The overall postoperative course was uneventful. The patient was discharged from our department on day 16 with very good swallow function and was decannulated after 1 week. Surgical resection was followed by postoperative radiation therapy (6400 cGy/32 fraction).\nThe final pathological report was of crucial importance in our case. First of all, the surgical margins of resection were found to be free of disease. Second, histological sections from the tumour of the glottis showed the characteristic morphology of squamous cell carcinoma. Cancer cells were large in size and polygonal in shape with eosinophilic cytoplasm and nuclei with moderate variation in size and shape. There were a moderate number of mitoses and keratinisation could be focally observed. Cancer cells showed an infiltrative pattern consisting mainly of nests and trabeculae that invaded the vocalis muscle in both the vocal cords. The perichondrium of thyroid cartilage was focally invaded by cancer cells. Histological sections from the tumour of the trachea showed morphological features identical to those of the tumour of the glottis. An upward infiltrating pattern could be noticed. Moreover, a comparative immunohistochemical study of the two tumours showed strong positivity of cancer cells in stains for keratins AE1/AE3 and 34βΕ12 and moderate positivity in stains for CK5/6, CK8/18 and epithelial membrane antigen. Immunohistochemistry for D2-40 antigen (podoplanin) illustrated the positivity of the lymphatic endothelium. Immunohistochemical stains for other vascular endothelia (CD31 and CD34 antigens) were also performed, and were negative. In the region between the two tumours, many lymphatics containing neoplastic emboli could be observed (). Finally, two tumour-infiltrated lymph nodes (the larger being of 1.2 cm diameter) with extracapsular spread were found in the left neck dissection specimen. A pT4a(m)N2b stage, according to eighth edition TNM staging, was established. |
Female xipho-omphalopagus conjoined twins were delivered by cesarean section at 36 weeks of gestation to a 29-year-old woman with a history of two pregnancies and one birth. The pregnant woman had no history of disease during the antenatal period. This patient was diagnosed with twins, but conjoined twins were unexpected. The conjoined twins were detected by ultrasound only when the pregnant woman was admitted to the hospital for delivery at a provincial hospital. Thus, the doctors decided to perform a cesarean section. The conjoined twins were safely delivered with a combined weight of 3.0 kg. The twins were joined at various organs, including the pericardial membrane, diaphragm, liver, and small intestine. In addition, baby A had a heart defect in the right chest and baby B had a hemangioma in the left arm. The twins were also joined at the upper abdomen from the sternum to umbilicus. Above and below the area of fusion, they showed two normal upper and lower limbs. The twins were placed under nursing care and followed up at the provincial hospital for 1.5 months. The twins were then referred to our hospital for continued follow-up examinations and a planned intervention. The twins were healthy and well-nourished. A multidisciplinary team that was formed in preparation for the intervention determined that separation should be delayed until the twins gained adequate weight with independent feeding and until they could tolerate the extended and intensive surgical procedure. It was thus suggested that surgical separation should proceed when the twins reached 10 months.\nThe twins presented with a combined weight of approximately 7 kg for surgical preparation (). The area of fusion from the sternum to the umbilicus was 18 cm in length and 6 cm in width. The right and left twins were marked as babies A and B, respectively (). The estimated skin and tissue surface area of 110 cm2 was considered to be deficient. In anticipation of insufficient skin coverage following twin separation, we determined that tissue expansion would be needed in order to allow closure. The area of fusion deviated from the midline, indicating that the twins’ anterior body had a larger area than the posterior body. A tissue expander was thus placed on the anterior body between the sternum and umbilicus because this placement fit the bottom portion of the tissue expander (). The tissue expander was a rectangular prism with dimensions of 7 cm (width) × 9.5 cm (length) × 5 cm (height), providing a theoretical volume of approximately 330 mL. A 4-cm skin incision was made parallel to, and approximately 2.5 cm from, one edge of the expander, on the fusion of the sternum ().\nA subcutaneous cavity with dimensions of 8 cm × 11 cm was made, and the tissue expander was placed between the subcutaneous and abdominal wall. We used three layers of nylon sutures to close the incision. A portion of the bottom of the expander contacted the twins’ ribs to avoid backward stretching during expansion. The injection port was then placed on baby B’s chest, and the expander was filled with a baseline of 70 mL sterile saline (0.9% NaCl) to stop blood from entering the dissection cavity. The expander was well-tolerated by the twins with no signs of local or systemic reactions. The first injection into the expander began on the 6th day of tissue expander insertion, before removing the sutures. We performed further injections into the expander every 2 days with approximately 30–70 mL per injection according to the expansion of the skin (). The expander reached 335 mL (or slightly higher than the expander’s theoretical volume) after six injections and within 10 days (). In order to prepare for surgical separation, expansion was completed on the 15th day after insertion. The expanded skin area was estimated at 180 cm2, which was sufficient to cover both patients’ skin deficiencies.\nThe twins presented for surgical separation 6 days following the end of tissue expansion. The surgical team was from our hospital. The total time for surgical separation and reconstruction was approximately 10 hours. The abdominal wall was closed in two layers, with the fascial muscle layer closed directly, using part of the fibrous layer around the expander, and the expanded skin of the chest was closed directly after the Z-lines incision. This allowed usage of the expanded skin at the ends of the expander. The sternal skin area was more challenging to close than the abdominal skin, so several additional incisions were made to enhance the tissue expander’s effect during closure. Within 72 hours after surgery, both babies were put on positive airway pressure and high-flow nasal cannulation. Both children had a good healing process on day 16 (), and they were discharged in good health 1 month after separation (). |
A 57-year-old female visited a respiratory internal physician due to suspected lung cancer (based on a mass screening chest X-ray examination). She did not have any symptoms. The chest X-ray showed a tumor shadow in the upper-middle field of the right lung with pleural effusion and a tumor shadow in the upper field of the left lung (). Computed tomography (CT) of the neck and chest revealed that the tumor shadows had been caused by a substernal goiter connected to the thyroid gland in the neck. According to the patient, she had been diagnosed with a goiter about 23 years ago, and it was followed up, but the follow-up process had been discontinued several times. After about 20 years, she visited our hospital for surgical treatment.\nIn a physical examination, the palpable thyroid gland was found to be diffusely swollen and soft and exhibited poor mobility. The lower pole of the thyroid was not palpable.\nA blood examination revealed normal thyroid function, a thyroglobulin level of 352 ng/ml, and negativity for the thyroglobulin antibody.\nUltrasound showed that the cervical thyroid gland was diffusely enlarged and exhibited multiple regions of cystic degeneration, but no obvious malignant findings were observed.\nCT of the neck and chest () showed the diffusely swollen thyroid gland and a substernal goiter, which extended to both sides of the thorax. Specifically, it extended to the bifurcation of the trachea on the dorsal side of the superior vena cava, the innominate vein, the aortic arch, and the ventral side of the trachea. The width of the goiter at the mediastinum was 145 mm (length: 80 mm, thickness: 80 mm). The right side of the substernal goiter was bigger than its left side. The interior of the lesion was heterogeneous, and calcification was seen in part of it. The goiter had compressed the trachea in the mediastinum, and the lumen of the trachea measured 6 mm in diameter at its narrowest point. Pleural effusion was noted in the right thorax. We performed 18F-fluorodeoxy glucose positron emission tomography to determine the malignancy of the substernal goiter, but no radiotracer accumulation was observed.\nWe also conducted a pathological examination. Fine-needle aspiration cytology of the cervical thyroid gland resulted in the lesion being classified as of “indeterminate significance,” and a pathological examination of a needle biopsy sample from the same site led to the lesion being diagnosed as a follicular neoplasm. Fine-needle aspiration cytology of the right pleural effusion demonstrated that it was benign.\nThe patient underwent total thyroidectomy using a transcervical and full sternotomy approach. The anesthesiologist intubated the patient with a bronchoscope. Although tracheal stenosis was observed, intubation was performed smoothly. Later, the tracheal tube was replaced with an NIM™ EMG endotracheal tube so that intraoperative nerve monitoring could be performed. The patient was placed in a supine position with her neck well extended. A cervical skin incision was made, and a median chest midline incision and full sternotomy were performed. First, we identified the bilateral vagal nerves and confirmed the absence of paralysis with the NIM™. As a preparation for the resection of the substernal goiter, the major blood vessels, including the innominate vein, brachiocephalic trunk, superior vena cava, and left subclavian artery, were carefully separated from the substernal goiter, and then thyroidectomy was performed ().\nThe right superior thyroid pedicle and right middle thyroid vein were ligated and dissected to allow the right thyroid lobe to be rotated to gain a view of the recurrent laryngeal nerve (RLN) from the lateral aspect of the thyroid gland, but the goiter prevented the right thyroid lobe from being rotated. It was difficult to identify the right RLN, so we decided to try to exteriorize the left thyroid lobe, which was smaller than the right thyroid lobe. The left superior thyroid pedicle and the left middle thyroid vein were ligated and dissected. The left thyroid lobe was more mobile than the right thyroid lobe, and the left RLN could be identified by rotating the left thyroid lobe in the medial direction. The NIM™ was effective at identifying the RLN. After identifying the left RLN, the left lower thyroid artery was ligated and dissected. The left RLN was carefully separated from the dorsal side of the left thyroid lobe and the substernal goiter so as not to cause any damage. The substernal goiter, which was connected to the left thyroid lobe, was pulled in the cranial direction, and the part adhering to the surrounding tissue, particularly the tissue between the goiter and the innominate vein, was dissected by ligation and coagulation with an energy device. Subsequently, the left thyroid lobe was also separated from the trachea. The exteriorization of the left thyroid lobe improved the mobility of the right thyroid lobe, and the right RLN was identified by dislocating the right upper pole to the caudal side. We carefully separated the right RLN from the goiter and ligated and dissected the right lower thyroid artery. We pulled the substernal portion of the right thyroid lobe gradually; separated the tissue connected to the goiter, including the left thyroid lobe; and succeeded in moving the substernal goiter in the cranial direction. The remaining attachments between the right thyroid lobe and trachea were broken, and a total thyroidectomy was conducted. We found three parathyroid glands had adhered to the resected thyroid gland, so we performed autotransplantation using the sternocleidomastoid muscle. The wound closed after drains were inserted in the neck and mediastinum. After the surgery, the patient was extubated immediately because no respiratory tract problems (e.g., tracheomalacia) were noted. The total duration of the operation was 9 h and 22 min, and the total amount of intraoperative blood loss was 3298 ml. The resected thyroid weighed 614 g ().\nPostoperative transient hypoparathyroidism was observed. Routine treatment with calcium (3 g daily orally) and 1 alpha-hydroxyvitamin D3 (2 μg daily orally) was administered. The patient was discharged home on the 9th postoperative day on levothyroxine (100 μg daily orally). A histopathological examination did not reveal any signs of malignancy, and so the lesion was diagnosed as an adenomatous goiter. |
A 54-year-old Caucasian male presented to our emergency department with severe epigastric pain with intractable nausea and vomiting over 48–72 hrs. The patient reported early satiety, a 30 lb. weight loss over 3 weeks and was unable to tolerate anything by mouth. Additionally, the patient had developed a left lateral neck mass which progressively increased in size over the past 9 months. Patient denied tenderness to palpation of the mass and dysphagia. His past medical history was significant for papillary carcinoma of the thyroid, GERD, chronic hepatitis C, polycystic kidney disease (PKD) and chronic kidney disease (CKD). His past surgical history was remarkable for thyroid malignancy treatment with radioactive iodine and total thyroidectomy in addition to a gunshot wound. Family and social history were not significant. On physical exam, a left supraclavicular soft tissue mass 6 cm in diameter was appreciated, it was non-tender with no submandibular lymphadenopathy. A soft tissue mass was also palpable on the anterior abdominal wall in the epigastric region.\nGiven the patient's malignant history, significant weight loss in the past 3 weeks with increasing left mass size, nausea, vomiting and abdominal pain, the patient was admitted for further evaluation. Initial studies included ultrasound of the abdomen for workup of acute pancreatitis with findings unremarkable for the body of the pancreas and the head and tail not being visualized. CT of the abdomen and pelvis revealed pulmonary nodules in the posterior segment of the right lower lobe of the lung and bronchiectasis of the left lower lobe, PKD and bilateral hepatic cysts. Oncology was consulted, and a CT of the chest and neck were ordered in addition to a chest X-ray that was remarkable for two pulmonary nodules with an enlarged paratracheal node. Pulmonology was consulted, and the patient was recommended to follow-up in 6 months for surveillance of the pulmonary nodules. ENT and Surgery were consulted, and an excisional lymph node biopsy was pursued due to suspicion of possible recurrence of previous thyroid malignancy or metastases (). Histopathology revealed metastatic mucin-producing adenocarcinoma. Immunoperoxidase studies performed on paraffin sections revealed the neoplastic cells were positive for CK20 and CDX2. The cells were negative for CK7, TTF-1, p63, prostate-specific antigen (PSA), prostatic acid phosphatase (PACP), chromogranin, synaptophysin and S-100. Gastroenterology was consulted, and an esophagogastroduodenoscopy was performed including an antral biopsy of the gastric mucosa, which was unremarkable. Colonoscopy of the lower GI tract revealed a 6 cm obstructing mass in the transverse colon and a biopsy was taken revealing findings of invasive well to moderately differentiated adenocarcinoma with mucinous features, focally present in subserosal tissue with ten of eleven regional lymph nodes involved. This confirmed the primary cancer originated from the colonic mass and metastasized to the left lateral neck mass (Virchow’s node).\nSurgical intervention was pursued with resection of the transverse colon with primary end-to-end anastomosis. This resolved the intractable acute abdomen. The patient was offered a trial of chemotherapy; however, given the patient’s diagnosis of metastatic colonic carcinoma, further treatment options were not pursued. Further discussion with the patient and his family led to a decision to proceed with comfort care measures. |
We present a 43-year-old woman with Raynaud’s syndrome secondary to lupus who was treated with bilateral popliteal nerve block catheters for ischemic pain and necrosis of her feet. This led to almost immediate relief of pain and return of color to her lower extremities. Prior to the procedure, the patient reported severe pain of 7–8 out of 10 on a numeric rating scale. The patient’s past medical history included severe mitral stenosis, aortic stenosis, hypertension, lupus nephritis, end-stage renal disease, and a third-degree heart block resulting in a pacemaker placement. The patient was noted to have systemic lupus erythematosus that was responsive only to steroids, after becoming refractory to prior treatments of Cytoxan and Imuran 11 years earlier. Our patient provided written consent for this case report and its publication. The Committee for the Protection of Human Rights of the University of Texas Health Science Center at Houston approved the retrospective review of this case.\nShe was originally admitted for dyspnea secondary to her aortic stenosis. The patient underwent open aortic valve and mitral valve replacement during her admission for her cardiac lesions. Her postoperative course was complicated by prolonged intubation and a flare-up of her lupus. She was followed by the rheumatology service for her lupus, who maintained her on prednisone 20 mg throughout her entire hospitalization so as to prevent a steroid withdrawal crisis. The cardiology service maintained her international normalized ratio goal at 2.5–3.5 as a result of her valve replacements. For relief of her lower extremity pain, the primary team prescribed amlodipine 10 mg by mouth daily and placed nitroglycerin patches 0.4 mg/h extended release on the soles of her feet in order to promote vasodilation and reduce cyanosis. When these measures proved ineffective, the Acute Pain Medicine Service was consulted.\nPhysical examination of our patient revealed an anxious woman in moderate distress. Her right lower extremity was cool to the touch from the toes to the ankle. There was dry gangrene on the second and third toes with dusky discoloration. Limitation of plantar flexion and dorsiflexion was observed secondary to pain and prolonged immobility. On the left foot, there was change in color with mottling of the skin but without significant dry gangrene. Limited range of motion was again seen secondary to pain. A complete blood count revealed white blood cell count of 7,500/mm3, hemoglobin 7.8 g/dL, and platelet 457,000/mm3. The patient was being anticoagulated with a heparin drip given her recent valve replacements. Her international normalized ratio at the time of the placement of the blocks was 3.21, which was within the target range of 2.5–3.5 desired by the cardiology service. Serum analysis for lupus was not done as the patient already had a confirmed diagnosis of lupus from a previous hospitalization. The patient repeatedly screamed in pain to even light touch of the lower legs or feet.\nWe elected to provide bilateral popliteal sciatic nerve block catheters for the purpose of reducing sympathetic innervation to her extremities, thereby promoting vasodilation. After informed consent was obtained from the patient, we administered 2 mg of midazolam intravenously for anxiety. The patient was placed in the supine position with her hip flexed and her left leg propped up on several blankets to allow access to her popliteal fossa. The ultrasound probe was placed in the popliteal fossa and scanned cephalad, paying close attention to the location where the sciatic nerve split into the tibial and common peroneal nerve branches. Using sterile technique, the skin was infiltrated with 2 cc of 1% lidocaine, and an 18 G 10 cm stimulating echogenic needle was inserted at the sciatic nerve under constant ultrasound visualization. A 20 mL bolus of 0.2% ropivacaine was given, and a 20 G catheter was inserted perineurally and secured at the skin. In a similar manner, the right sciatic nerve block catheter was placed without complication. The catheters were then connected to two distinct computer-assisted drug delivery pumps, one on each side, with 0.2% ropivacaine infusion at 6 mL/h without the option for patient-controlled analgesia boluses.\nWithin 30 minutes of completion of the procedure, there was a reddish hue to the previously dusky areas indicative of reperfusion to her toes. She was able to stand on her feet within 24 hours, with the assistance from a physical therapist. On day 2, the catheters were removed and the infusions discontinued. The patient reported that she was able to ambulate on her feet from the second day with complete resolution of the pain in the remainder of her hospital stay. Cyanosis of the skin also improved significantly during her hospital stay. There was no hematoma, bleeding, or other complications noted. Further follow-up revealed that several months later, the patient underwent elective partial amputation of her toes bilaterally, which had been noted to have dry gangrene prior to the initiation of the peripheral nerve blocks. |
A 74-year-old woman was referred to our clinic for DMD of the left eye. She underwent uncomplicated phacoemulsification cataract surgery with a temporal corneal incision at a local eye clinic one week before referral to our clinic. She had received cataract surgery in her right eye four years earlier at another clinic. She had no remarkable medical history or ocular trauma history. She had an intracameral air injection at the local clinic four days after the surgery on her left eye due to the DMD. On examination in our clinic, her uncorrected visual acuity was counting fingers at 30 cm in her left eye, and her intraocular pressure was 17 mmHg. Slit lamp examination showed extensive DMD with only a small portion of attached endothelium in the center of the cornea (). The next day, an intracameral gas injection of 14% C3F8 was performed on the left eye in the operating room. The interface fluid between the Descemet's membrane and the stroma was removed using an ab externo stab incision of the corneal stroma in mid-periphery (). The patient was instructed to maintain a supine position. One day after the gas injection, her uncorrected visual acuity was counting fingers at 30 cm, and her intraocular pressure was 10 mmHg. Only a small area of detachment remained in the inferior cornea. The patient was hospitalized for close follow-up. Two days after the gas injection, her uncorrected visual acuity was 20/400, her intraocular pressure was 15 mmHg, and a small area of detachment was still visible. The postoperative medications of ofloxacin eye drops and 0.12% prednisolone eye drops were maintained every two hours. Three days after the gas injection, the detachment had resolved completely, but the intraocular pressure was elevated to 52 mmHg. The gas in the anterior chamber had increased and some gas could also be observed in the posterior chamber, resulting in an iris bombe. To reduce intraocular pressure after mannitolization, we prescribed Timolol (Timoptic XE®) eye drops once daily, atropine 1% eye drops three times daily, and oral acetazolamide 500 mg divided into four doses. Four days following the gas injection, her intraocular pressure was 7 mmHg, and the gas bubble was still visible in the anterior chamber. The Descemet's membrane remained well-attached. Eight days after the gas injection, her corrected vision was 20/30 and her intraocular pressure was 6 mmHg. Two weeks after the gas injection, the attached Descemet's membrane appeared stable and the size of the gas bubble had decreased to 25% of the vertical chamber height. One month following the procedure, the patient's corrected visual acuity remained stable at 20/30, and the intraocular pressure was 13 mmHg. The gas bubble in the anterior chamber decreased significantly, to only a small bubble in the superior portion. |
A 34-year-old woman presented to the emergency room with a few days history of dizziness and lethargy. Severe anemia was detected (Hb 54 g/L, MCV 79 fl). The patient had not experienced hematemesis or fresh blood in stool but she had experienced an isolated incident of diarrhea, dark in color, two days before presenting to the hospital and at least one time before in the same month. Three weeks earlier she had been admitted to the hospital with the same symptoms and was found to have iron-deficiency anemia. Upper gastrointestinal endoscopy and colonoscopy did not reveal any explanation for the anemia. She was discharged, after receiving a blood transfusion, with a planned capsular endoscopy (CE) but presented to the hospital again with worsening symptoms before the CE was performed.\nHer past medical history was unremarkable except for hypothyroidism and her only regular medication was the thyroxin replacement. A year earlier she had suffered a motor vehicle accident with a rather serious frontal collision, but examination at the emergency reception did not indicate need for computerized tomography (CT) scan to be undertaken at the time. She was a nonsmoker and used alcohol in moderate amounts.\nAt the current presentation physical examination revealed tachycardia (126 beats per min) but normal blood pressure (117/66 mmHg) and otherwise normal vital signs. Examination of the abdomen revealed no tenderness, abnormal masses, or organomegalies. Apart from the marked microcytic anemia, other blood tests were within normal range, including liver tests. A slight elevation of the tumor marker CA 19-9 (41.2 U/mL) was found. An upper gastrointestinal endoscopy revealed isolated gastric varices in the fundus of the stomach and red spots in the mucosa of the varices indicating a previous recent bleeding (). However no active bleeding was seen and no blood was visible in the stomach. These gastric varices had not been observed in the prior endoscopy, performed three weeks earlier. An abdominal CT was performed in order to identify the underlying pathology, mainly to look for portal vein thrombosis which was the most important differential diagnosis since the patient's liver tests were normal. The images showed a large cystic tumor, 10.4 × 11.4 cm in diameter, located in the tail of the pancreas (). Moderate splenomegaly (16 cm) was also detected on the CT.\nLaparotomy demonstrated that the tumor had massive adhesions to surrounding organs. In order to resect the tumor a distal pancreatectomy was performed along with a splenectomy, a sleeve resection of the stomach, and a partial hemicolectomy. Macroscopic pathological examination displayed a multilocular cystic tumor, 12 cm in greatest dimension with one large dominating cyst and multiple smaller ones, containing yellowish fluid and no papillary structures on the internal surface. The cyst walls were fibrous and were attached to the surrounding organs. Microscopic examination showed a mucinous, columnar epithelium lining the cystic surfaces, without any cellular or nuclear atypia. The subepithelial stroma was of ovarian-type. These findings were compatible with the final diagnosis of mucinous cystadenoma (). Given the previous history of a motor vehicle accident a year earlier, it is conceivable that a traumatic rupture of the cyst has occurred with secondary scar tissue development. The patient had uneventful and full recovery. |
An 85-year-old Caucasian male was admitted after sudden onset of expressive aphasia and weakness in both legs lasting 20 seconds. He was athletic, self-reliant and had no cognitive impairment. During the last 28 years, he had experienced 8-10 heterogeneous episodes of acute neurological symptoms, such as central facial palsy, hemiparesis, and non-fluent aphasia, lasting from seconds to 3-4 hours. Precerebral duplex and electrocardiography (ECG) were performed several times with normal results, and EEG registration and 24-hour Holter monitoring had been normal. Previous MRI scans showed no abnormal restricted diffusion, as seen in acute cerebral infarcts, but infarct sequelae in the left temporal lobe and both thalami. Several years later, three additional infarct sequelae were detected in the cerebellum. The patient was treated with platelet inhibitors, and medications and dosages were adjusted after new episodes. There was no suspicion of lack of compliance. Except from age, migraine, and previous smoking, with cessation 35 years ago, he had no known risk factors for cerebrovascular disease. On the current admission, he presented with reduced motor speed in his left arm and leg. Electrocardiography and Holter monitoring showed no signs of atrial fibrillation. CT and MRI revealed multiple, cortical infarct sequelae in the anterior and posterior circulation territories of both hemispheres, and MRI also detected two acute embolic infarcts in the right occipital lobe and one in the left parietal lobe (Figure ). CT and MRI angiograms and duplex sonography did not show significant plaques or stenoses, and pre- and intracerebral flow were normal with asymmetrical vertebral arteries, which were considered a normal anatomical variant. Cortical infarcts in several vascular territories strongly suggest cardioembolic etiology, but transthoracic echocardiogram showed no cardiac sources of emboli, and there was no sign of left atrial enlargement, which may be seen in the presence of atrial fibrillation. The patient concurred to further diagnostic tests aiming to determine the cause of recurrent cerebral emboli, although he was informed that the results would not necessarily alter treatment recommendations. We performed a transcranial Doppler (TCD) bubble test with 10 mL air-mixed saline injected into the left cubital vein while the left middle cerebral artery was insonated with a 2-MHz probe. Injection at resting state produced no microembolic signals, while injection after Valsalva maneuver resulted in a shower of microembolic signals followed by single signals persisting for over 30 seconds. The result implied the presence of a latent right-to-left shunt, and transesophageal echocardiography verified a large patent foramen ovale (PFO; Figure ). In agreement with the patient, we decided on non-operative treatment. Due to previous failure of antiplatelet treatment, we changed to a direct oral anticoagulant (dabigatran 110 mg twice daily), intended as a lifelong treatment. He had no subjective complaints at discharge. |
A 57-year-old female visited a respiratory internal physician due to suspected lung cancer (based on a mass screening chest X-ray examination). She did not have any symptoms. The chest X-ray showed a tumor shadow in the upper-middle field of the right lung with pleural effusion and a tumor shadow in the upper field of the left lung (). Computed tomography (CT) of the neck and chest revealed that the tumor shadows had been caused by a substernal goiter connected to the thyroid gland in the neck. According to the patient, she had been diagnosed with a goiter about 23 years ago, and it was followed up, but the follow-up process had been discontinued several times. After about 20 years, she visited our hospital for surgical treatment.\nIn a physical examination, the palpable thyroid gland was found to be diffusely swollen and soft and exhibited poor mobility. The lower pole of the thyroid was not palpable.\nA blood examination revealed normal thyroid function, a thyroglobulin level of 352 ng/ml, and negativity for the thyroglobulin antibody.\nUltrasound showed that the cervical thyroid gland was diffusely enlarged and exhibited multiple regions of cystic degeneration, but no obvious malignant findings were observed.\nCT of the neck and chest () showed the diffusely swollen thyroid gland and a substernal goiter, which extended to both sides of the thorax. Specifically, it extended to the bifurcation of the trachea on the dorsal side of the superior vena cava, the innominate vein, the aortic arch, and the ventral side of the trachea. The width of the goiter at the mediastinum was 145 mm (length: 80 mm, thickness: 80 mm). The right side of the substernal goiter was bigger than its left side. The interior of the lesion was heterogeneous, and calcification was seen in part of it. The goiter had compressed the trachea in the mediastinum, and the lumen of the trachea measured 6 mm in diameter at its narrowest point. Pleural effusion was noted in the right thorax. We performed 18F-fluorodeoxy glucose positron emission tomography to determine the malignancy of the substernal goiter, but no radiotracer accumulation was observed.\nWe also conducted a pathological examination. Fine-needle aspiration cytology of the cervical thyroid gland resulted in the lesion being classified as of “indeterminate significance,” and a pathological examination of a needle biopsy sample from the same site led to the lesion being diagnosed as a follicular neoplasm. Fine-needle aspiration cytology of the right pleural effusion demonstrated that it was benign.\nThe patient underwent total thyroidectomy using a transcervical and full sternotomy approach. The anesthesiologist intubated the patient with a bronchoscope. Although tracheal stenosis was observed, intubation was performed smoothly. Later, the tracheal tube was replaced with an NIM™ EMG endotracheal tube so that intraoperative nerve monitoring could be performed. The patient was placed in a supine position with her neck well extended. A cervical skin incision was made, and a median chest midline incision and full sternotomy were performed. First, we identified the bilateral vagal nerves and confirmed the absence of paralysis with the NIM™. As a preparation for the resection of the substernal goiter, the major blood vessels, including the innominate vein, brachiocephalic trunk, superior vena cava, and left subclavian artery, were carefully separated from the substernal goiter, and then thyroidectomy was performed ().\nThe right superior thyroid pedicle and right middle thyroid vein were ligated and dissected to allow the right thyroid lobe to be rotated to gain a view of the recurrent laryngeal nerve (RLN) from the lateral aspect of the thyroid gland, but the goiter prevented the right thyroid lobe from being rotated. It was difficult to identify the right RLN, so we decided to try to exteriorize the left thyroid lobe, which was smaller than the right thyroid lobe. The left superior thyroid pedicle and the left middle thyroid vein were ligated and dissected. The left thyroid lobe was more mobile than the right thyroid lobe, and the left RLN could be identified by rotating the left thyroid lobe in the medial direction. The NIM™ was effective at identifying the RLN. After identifying the left RLN, the left lower thyroid artery was ligated and dissected. The left RLN was carefully separated from the dorsal side of the left thyroid lobe and the substernal goiter so as not to cause any damage. The substernal goiter, which was connected to the left thyroid lobe, was pulled in the cranial direction, and the part adhering to the surrounding tissue, particularly the tissue between the goiter and the innominate vein, was dissected by ligation and coagulation with an energy device. Subsequently, the left thyroid lobe was also separated from the trachea. The exteriorization of the left thyroid lobe improved the mobility of the right thyroid lobe, and the right RLN was identified by dislocating the right upper pole to the caudal side. We carefully separated the right RLN from the goiter and ligated and dissected the right lower thyroid artery. We pulled the substernal portion of the right thyroid lobe gradually; separated the tissue connected to the goiter, including the left thyroid lobe; and succeeded in moving the substernal goiter in the cranial direction. The remaining attachments between the right thyroid lobe and trachea were broken, and a total thyroidectomy was conducted. We found three parathyroid glands had adhered to the resected thyroid gland, so we performed autotransplantation using the sternocleidomastoid muscle. The wound closed after drains were inserted in the neck and mediastinum. After the surgery, the patient was extubated immediately because no respiratory tract problems (e.g., tracheomalacia) were noted. The total duration of the operation was 9 h and 22 min, and the total amount of intraoperative blood loss was 3298 ml. The resected thyroid weighed 614 g ().\nPostoperative transient hypoparathyroidism was observed. Routine treatment with calcium (3 g daily orally) and 1 alpha-hydroxyvitamin D3 (2 μg daily orally) was administered. The patient was discharged home on the 9th postoperative day on levothyroxine (100 μg daily orally). A histopathological examination did not reveal any signs of malignancy, and so the lesion was diagnosed as an adenomatous goiter. |
We describe the case of a 7-year-old male who presented with a first episode of arterial ischemic stroke. The neurological evaluation revealed a left sided hemiparesis which was not involving the facial musculature. The initial CT scan revealed an area of hypointensity in the region of the right basal ganglia. More specifically, its anatomical contribution was extending to the anterior limb and the genu of the right internal capsule, and in the nearby territory of the globus pallidus. This infarct refers to the anatomical contribution of the medial and lateral lenticulostriate arteries and was attributed possibly due to the migration of an arterial thrombus to their vessel of origin (–).\nThe diagnostic work up for the recognition of the underlying cause of the AIS included the investigation of possible prothrombotic conditions and thrombophilia, but it did not reveal any underlying pathologic condition. The patient underwent a detailed cardiologic work up, which included a transesophageal echocardiographic examination. It revealed the presence of a patent foramen ovale (capillary subtype) and a patent ductus arteriosus (, ).\nBased on these findings, the patient was immediately treated with anticoagulation therapy. Despite that, he developed a second episode of arterial ischemic stroke 9 months later. The neurological evaluation after the second ictus revealed complete left-sided hemiparesis with involvement of the musculature of the ipsilateral half of the face.\nHe underwent a radiological evaluation with a new CT scan, which revealed an extensive area of hypointensity, extending to the mesencephalon-pontine region the dimensions of the basal ganglia infarction were significantly reduced. An additional new infarction territory was recognized, in the area of the right cerebellar hemisphere, lateral to the vermis. The anatomic substrate of the new stroke could be attributed to a second stroke that developed in the region that is nourished from the perforators of the basal artery, near its terminal bifurcation. The infarct with a hemispheric distribution was attributed to a thrombus-related obstruction of hemispheric branches of the vertebrobasilar arterial system (–).\nAfter the second episode of AIS, we decided to occlude the foramen ovale with the use of the Amplatzer PFO Occluder Cribiform No 18, and by the way, of the patent ductus arteriosus with an MReye Flipper PDA Closure Detachable Coil IMWCE-8-PDA-4 Coil 8 mm.\nWe performed retrograde catheterization of the ascending aorta through the right femoral artery, utilizing the percutaneous technique. Through the patent ductus arteriosus, the catheter was inserted to the pulmonary artery. Insertion and detachment of the detachable coil from the arterial route followed (, ). Next step was the catheterization of the right cardiac cavities and of the pulmonary artery via the right femoral vein, using the percutaneous technique. The catheter is inserted into the left atrium and the left superior pulmonary vein, via the PFO. The system responsible for deployment of the Amplatzer PFO Ocluder was inserted via the transvenous route, and the ocluder was detached (, ), (). After the procedure, a gradual discontinuation of anticoagulant therapy was decided, which was followed by replacement with antiplatelet therapy, namely aspirin.\nThe follow-up period extends to approximately one and a half years, during which neither adverse effects from the therapy were noted, nor any new ischemic strokes. The patient’s neurological status remains stable. |
A 71-year-old man with a history of TEVAR for Stanford B aortic dissection and aortic aneurysm rupture 20 months ago presented at a local medical clinic with fever over 38 °C. Laboratory findings revealed elevated infectious indicators, and he was prescribed with antibiotics. However, he presented at his primary care hospital one week later without symptomatic improvement. Contrast-enhanced computed tomography at that time identified a fistula between the esophagus (A and B) and an aortic aneurysm, and upper gastrointestinal endoscopy revealed an esophageal ulcer (C). Therefore, he was diagnosed with AEF after TEVAR and immediately transferred to our hospital for surgical therapy. On admission, he did not have hematemesis and was hemodynamically stable. He required emergency surgery to the control the spread of infection and prevent fatal bleeding. We planned a three-step surgical approach ().\nThe first step of the procedure on the day of admission comprised esophagectomy via a right thoracotomy at the fourth intercostal space with the patient in the left lateral position. We cut the esophagus above the AEF and diaphragm and resected part of it that also included the AEF. Intraoperative findings revealed extensive inflammation of the mediastinal tissue and leakage of infected old blood from the aortic fistula without massive bleeding (A). We placed drains in the right thoracic cavity and the mediastinum beside the aortic fistula. The patient was then placed in the supine position and the residual esophagus was brought to the left cervical region as an esophagostomy. A feeding jejunostomy tube was then placed via a small abdominal incision. He was admitted to the surgical intensive care unit thereafter, and infection control was started by abscess draining and antibiotic administration. Gross findings of the resected esophagus showed a perforation site with a maximum diameter of 1.0 cm (B).\nNo bacteria were identified in blood culture; however, Klebsiella pneumonia and Prevotella melaninogenic were identified in mediastinal tissue culture. Antibiotics, abscess drainage and pleural lavage were performed, however they did not completely improve the inflammatory response after the first surgery. Therefore, we removed residual infected foci as soon as the patient’s status became stabilized. The second step of the procedure was implemented one month later to remove the thoracic aortic aneurysm and artificial stent-graft and to restore the aorta in situ with a synthetic vascular prosthesis through a left thoracotomy. The prosthesis was infiltrated with rifampicin before graft replacement to prevent repeated infection. Thereafter, the inflammatory response and the general status of the patient gradually improved under antibiotics, drainage and pleural lavage. He developed strength through postoperative rehabilitation and enteral nutrition management.\nThree months after the second step, the third step addressed the esophageal defect. A narrow gastric tube fashioned by laparotomy was brought up through the ante-thoracic route, and cervical esophagogastrostomy proceeded. The patient recovered uneventfully, resumed oral intake and was discharged on postoperative day 37. The patient remains free of disease and adverse events at 24 months after completing the three-step procedure. |
A 42-yr-old male was referred to Ulsan University Hospital for evaluation of a multinodular mass based on the left diaphragmatic pleura. This mass had been detected incidentally on a chest radiograph during the patient's annual medical checkup. The patient had been in good general condition, except for left inferior pleural thickening on chest radiographs taken from 1999 to 2004. A newly developed small nodule was detected in 2005, but the clinician regarded it as an inactive lesion. In 2007, the lesion had enlarged and formed a sizable mass, suggesting a malignancy (). The patient had smoked one-half to one pack of cigarettes per day for 14 yr. On admission, all laboratory data were within normal ranges. Computed tomography (CT) revealed a homogeneously well-enhanced multinodular mass, measuring 4.8 cm in its greatest dimension, based on the diaphragm (). The patient was suspected of having a tumor of pleural origin such as a sclerosing hemangioma. There was no evidence of a lung parenchymal mass or a primary malignancy of other organs. An excision by VATS was attempted.\nIntraoperatively, the mass had the gross appearance of a hemangioma containing blood. The mass, which was tightly attached to the diaphragm and adjacent lung tissue, was removed through a minimal thoracotomy. A piece of soft tissue labeled "diaphragm" was submitted for a frozen section, which revealed a multinodular solid mass containing abundant lymphoid cells in a diffuse or vaguely nodular arrangement, as well as a thick fibrous capsule and septae, suggesting it may be a low grade malignant lymphoma (). The entire mass and the attached lung tissue were excised. Examination of a permanent section showed that the mass was ectopic splenic tissue, which included white and red pulp and central arteries ().\nA review of the patient's medical history showed that, 20 yr earlier, he had been pierced through the left abdomen and back by an iron bar. The patient, however, did not know what kind of operation had been performed. Review of his chest CT at admission revealed radiologic evidence that he had undergone a splenectomy. |
A 58-year-old female presented to infectious disease department of the hospital with a severe pulmonary infection on the background of morbid obesity (BMI 43) and type 2 diabetes mellitus for the last 17 years which was under treatment with insulin in addition to hypertension and hyperlipidaemia. She has past surgical history of cholecystectomy. Recently she has lost 25 kg using Liraglutide (Victoza) injections. She weighed 110 kg at the time of admission.\nShe has no urological symptoms; no flank pain, dysuria, or frequency or gross haematuria, and neither had she reported experiencing any of these symptoms in the past. She had an elevated ESR for a long time with unknown cause. Urine analysis revealed microscopic haematuria. Besides treatment for pulmonary infection, work-up has been initiated to find the cause of elevated ESR and microscopic haematuria.\nAn ultrasonography of abdomen and pelvis was performed which revealed a large round hypoechoic solid appearing mass at the lower pole of the right kidney. The mass is virtually indistinguishable from a renal malignancy. The ultrasonography of her abdomen and pelvis was otherwise unremarkable except for evidence of previous cholecystectomy and two small lesions in the liver suspected to be hemangiomas. Then abdominal Computed Tomography with and without administration of contrast medium injection was performed to further investigate the lesion visualized by ultrasonography (). A 10 cm × 8 cm heterogeneous soft tissue mass in the lower pole of the right kidney was reported. The mass had faint enhancement and adjacent fatty stranding and pararenal facial thickening.\nAlso, bear's paw sign was observed due to dilation of the renal calyces on CT of the abdomen (). Complex cystic renal mass or renal malignancy and cystic degeneration were mentioned as a probable diagnosis.\nIn addition, chest X-ray revealed mild pleural effusion and a soft tissue density pleural based lesion in right hemithorax. A thoracic CT scan with and without contrast medium injection was advised to investigate the latter findings further ().\nThoracic CT revealed bilateral smaller than 2 cm irregular bordered nodules in both lung fields. Also, a 3.5 cm × 2.5 cm cavitating lesion in the right upper lobe, a wedge-shaped consolidation in the right lower lobe, right pleural effusion, and right hilar adenopathy were seen in thoracic CT scan. Thoracic metastasis was suggested.\nOn laboratory investigations before treatment, other than elevated ESR (99 in 1 hour), urinalysis shows protein, glucose, and blood in a turbid sample. Urine culture was negative; therefore antibiotics were not administered. Biochemistry shows low sodium (130 normal range: 136–145). Complete blood count shows normocytic anaemia (Hb: 10.3 normal range: 12–17) and slightly elevated white cell count (10.1 normal range 4–10). Tumour markers, CEA, CA19-9, CA125, and CA15-3, were not elevated.\nWith regard to the imaging findings, renal malignancy () with pulmonary metastasis () was suggested. After consultation with the urologist, the patient was scheduled for right radical nephrectomy. In semiflank position (mild elevated patient right flank), with transperitoneal subcostal incision, classic right radical nephrectomy was performed. Nephrectomy is recommended in patients with an irretrievably impaired kidney due to symptomatic, chronic infection, calculus disease, or severe traumatic injury []. The mass had severe adhesions to adjacent organs which were released during operation. The entire mass was sent for pathologic examination.\nAfter operation (right nephrectomy) the patient is in good condition without fever and pulmonary symptoms. Also, ESR is reduced to 65 mm/hr. White cell count was reduced to 7.1 and is within the normal range. It seems that her pulmonary nodules had been septic pulmonary embolisms. There is a similar case presentation in the literature which reports XGP complicated with pulmonary embolism []. Our case is the second presenting with this complication.\nOn pathological examination, Xanthogranulomatous pyelonephritis (XGP) was diagnosed. On microscopic examination, there is the focal replacement of renal parenchyma by severe mixed inflammatory cells infiltration including lymphoplasma cells, neutrophils and foamy histiocytes infiltration, and fibrosis with extension to perirenal soft tissue. Diabetic nephropathy including nodular sclerosis and arteriolar hyalinosis is seen in the background ().\nProphylactic antibiotics were administered at surgery. About three months following right nephrectomy, the pulmonary nodules were found to have spontaneously resolved on chest CT scan. |
A 57-year-old female visited a respiratory internal physician due to suspected lung cancer (based on a mass screening chest X-ray examination). She did not have any symptoms. The chest X-ray showed a tumor shadow in the upper-middle field of the right lung with pleural effusion and a tumor shadow in the upper field of the left lung (). Computed tomography (CT) of the neck and chest revealed that the tumor shadows had been caused by a substernal goiter connected to the thyroid gland in the neck. According to the patient, she had been diagnosed with a goiter about 23 years ago, and it was followed up, but the follow-up process had been discontinued several times. After about 20 years, she visited our hospital for surgical treatment.\nIn a physical examination, the palpable thyroid gland was found to be diffusely swollen and soft and exhibited poor mobility. The lower pole of the thyroid was not palpable.\nA blood examination revealed normal thyroid function, a thyroglobulin level of 352 ng/ml, and negativity for the thyroglobulin antibody.\nUltrasound showed that the cervical thyroid gland was diffusely enlarged and exhibited multiple regions of cystic degeneration, but no obvious malignant findings were observed.\nCT of the neck and chest () showed the diffusely swollen thyroid gland and a substernal goiter, which extended to both sides of the thorax. Specifically, it extended to the bifurcation of the trachea on the dorsal side of the superior vena cava, the innominate vein, the aortic arch, and the ventral side of the trachea. The width of the goiter at the mediastinum was 145 mm (length: 80 mm, thickness: 80 mm). The right side of the substernal goiter was bigger than its left side. The interior of the lesion was heterogeneous, and calcification was seen in part of it. The goiter had compressed the trachea in the mediastinum, and the lumen of the trachea measured 6 mm in diameter at its narrowest point. Pleural effusion was noted in the right thorax. We performed 18F-fluorodeoxy glucose positron emission tomography to determine the malignancy of the substernal goiter, but no radiotracer accumulation was observed.\nWe also conducted a pathological examination. Fine-needle aspiration cytology of the cervical thyroid gland resulted in the lesion being classified as of “indeterminate significance,” and a pathological examination of a needle biopsy sample from the same site led to the lesion being diagnosed as a follicular neoplasm. Fine-needle aspiration cytology of the right pleural effusion demonstrated that it was benign.\nThe patient underwent total thyroidectomy using a transcervical and full sternotomy approach. The anesthesiologist intubated the patient with a bronchoscope. Although tracheal stenosis was observed, intubation was performed smoothly. Later, the tracheal tube was replaced with an NIM™ EMG endotracheal tube so that intraoperative nerve monitoring could be performed. The patient was placed in a supine position with her neck well extended. A cervical skin incision was made, and a median chest midline incision and full sternotomy were performed. First, we identified the bilateral vagal nerves and confirmed the absence of paralysis with the NIM™. As a preparation for the resection of the substernal goiter, the major blood vessels, including the innominate vein, brachiocephalic trunk, superior vena cava, and left subclavian artery, were carefully separated from the substernal goiter, and then thyroidectomy was performed ().\nThe right superior thyroid pedicle and right middle thyroid vein were ligated and dissected to allow the right thyroid lobe to be rotated to gain a view of the recurrent laryngeal nerve (RLN) from the lateral aspect of the thyroid gland, but the goiter prevented the right thyroid lobe from being rotated. It was difficult to identify the right RLN, so we decided to try to exteriorize the left thyroid lobe, which was smaller than the right thyroid lobe. The left superior thyroid pedicle and the left middle thyroid vein were ligated and dissected. The left thyroid lobe was more mobile than the right thyroid lobe, and the left RLN could be identified by rotating the left thyroid lobe in the medial direction. The NIM™ was effective at identifying the RLN. After identifying the left RLN, the left lower thyroid artery was ligated and dissected. The left RLN was carefully separated from the dorsal side of the left thyroid lobe and the substernal goiter so as not to cause any damage. The substernal goiter, which was connected to the left thyroid lobe, was pulled in the cranial direction, and the part adhering to the surrounding tissue, particularly the tissue between the goiter and the innominate vein, was dissected by ligation and coagulation with an energy device. Subsequently, the left thyroid lobe was also separated from the trachea. The exteriorization of the left thyroid lobe improved the mobility of the right thyroid lobe, and the right RLN was identified by dislocating the right upper pole to the caudal side. We carefully separated the right RLN from the goiter and ligated and dissected the right lower thyroid artery. We pulled the substernal portion of the right thyroid lobe gradually; separated the tissue connected to the goiter, including the left thyroid lobe; and succeeded in moving the substernal goiter in the cranial direction. The remaining attachments between the right thyroid lobe and trachea were broken, and a total thyroidectomy was conducted. We found three parathyroid glands had adhered to the resected thyroid gland, so we performed autotransplantation using the sternocleidomastoid muscle. The wound closed after drains were inserted in the neck and mediastinum. After the surgery, the patient was extubated immediately because no respiratory tract problems (e.g., tracheomalacia) were noted. The total duration of the operation was 9 h and 22 min, and the total amount of intraoperative blood loss was 3298 ml. The resected thyroid weighed 614 g ().\nPostoperative transient hypoparathyroidism was observed. Routine treatment with calcium (3 g daily orally) and 1 alpha-hydroxyvitamin D3 (2 μg daily orally) was administered. The patient was discharged home on the 9th postoperative day on levothyroxine (100 μg daily orally). A histopathological examination did not reveal any signs of malignancy, and so the lesion was diagnosed as an adenomatous goiter. |
A 14-year-old boy presented to us with a 2 weeks old infected bimalleolar defect with dimensions of 12 cm × 7 cm medially and 8 cm × 4 cm laterally [] at the ankle. The patient was treated with cast application elsewhere for swelling of the ankle following a fall and had no bony injury. He developed severe pain in the ankle soon after and fever a week later,. On removal of the cast, necrosis of the skin was noted over the both malleoli which was debrided at the referral hospital and then he was referred to our institute for further management. Malleoli and the adjacent lower ends of tibia and fibula were exposed with open ankle joint-draining frank pus. The foot was sensate and well vascularised with good dorsalis pedis and posterior tibial pulsations. After containment of purulent discharge, flap cover was planned. Considering the dimensions and proximity of the two defects, a chimeric fasciocutanous anteromedial thigh (AMT) and anterolateral thigh (ALT) flap [] based on the lateral circumflex, femoral vessels was planned and their perforators marked with hand held Doppler. The flaps were raised based on these perforators and the pedicles traced proximally for their confluence. It was found to be very close to the origin of the lateral circumflex femoral artery precluding chimeric flap design and due to gross vessel size mismatch and excess pedicle length. The second team explored the anterior tibial artery and both vena comitans. The vessels were healthy and amenable for the use with good pulsatile flow from both the proximal and distal cut ends of the recipient artery. Both the flaps were harvested independently with adequate pedicle length. Both flap vessels were anstomosed to the proximal and distal cut ends of the anterior tibial artery, in an end to end fashion. The corresponding flap veins were anastomosed to the available two vena comitans of the anterior tibial artery in an antegrade direction. Both the flaps survived without any complications []. At 11 months follow-up, the patient is ambulatory with a stiff ankle in slight varus position with X-ray showing extensive post-septic arthritic sequale of the ankle joint []. |
Case 2. A 70-year-old man was investigated 3 years earlier when he presented with visible haematuria urinary frequency and occasional straining to void. His repeated urine cytology and intravenous urography were normal and his cystoscopic examination revealed a focal area of localized inflammation on the floor of the urinary bladder lateral to the right ureteric orifice. He was lost to follow-up. However, 3 years later, he presented with recurrent visible haematuria. He had a computed tomography scan which revealed a mild-to-moderate dilation of the distal part of the right ureter and 0.8 cm thickening of the wall of the urinary bladder around the right ureteric orifice. There was no evidence of any significant lymph adenopathy or any other abnormal mass in the pelvis. He underwent transurethral biopsy of the thickened region and histological examination of the specimen revealed infiltration of lamina propria by ill-defined nested neoplastic cells with bland cytologic characteristics (see ). There was also evidence of perineural invasion by the tumour. The immunohistochemical staining characteristics of the tumour include immunoreactivity for high-molecular cytokeratin 903 and cytokeratin 7 and negativity for prostate specific antigen, prostate acid phosphatase, S100, and chromogranin. He subsequently underwent radical cystoprostatectomy elsewhere. The histology report from the institution where he underwent radical cystoprostatectomy stated that the tumour had extended through the lateral wall of the right trigone with vascular invasion and a focus of urothelial carcinoma in situ. The histology report also stated that in addition to the urinary bladder tumour there was focal adenocarcinoma of the prostate gland (Gleason pattern 3 + 3 = 6) which was confined to the left lobe of the prostate gland. The perivesical lymph nodes were negative for metastatic disease. This patient was lost to follow-up two months pursuant to his surgery.\nXiao and associates [] stated that additional immunohistochemical stainings were done on the tumour specimens of the two patients and these showed that the nested neoplastic cells in both cases were strongly immunoreactive for p63 (a homolog of p53 protein) (Figures and ); 40% to 50% and 30% to 40% of tumour cells in Case 1 exhibited strong positivity for p53 and Ki-67, respectively, and no staining difference for either p53 or Ki-67 was present between superficial and deep infiltrating tumour cells. Focally positive stains for both p53 and Ki-67 were exhibited in the biopsy specimen of Case 2. The experience learnt from the biological behaviour of both Cases 1 and 2, especially in Case 1, would be indicative of the aggressive nature of nested variant of urothelial carcinoma.\nKrishnamoorthy and associates [] reported the case of a 46-year-old woman who presented with a two-year history of interrupted stream of urine on voiding. She developed acute urinary retention for which she was catheterized. Further evaluation after her catheterization revealed a large urinary bladder tumour. She did not have any history of haematuria or urinary tract infection. Her urine microscopy revealed 15 red blood cells per high power field. She had ultrasound scan of the abdomen which revealed bilateral mild hydroureteronephrosis up to the vesicoureteric junction. A huge heteroechoic pedunculated mass which measured 10 cm in size, with smooth surface and well-defined margin in the bladder, occupying the entire surface of the urinary bladder. She also had a contrast computed tomography scan of the abdomen which revealed normal liver, pancreas, and adrenals. The computed tomography scan also showed bilateral mild hydroureteronephrosis with a 10 mm simple cyst in the interpolar region of the right kidney. There was a large heterodense mass in the urinary bladder which occupied most of the bladder. There were multiple enlarged lymph nodes involving the parailiac right external iliac, left internal iliac, and left inguinal regions, each measuring from 7 mm to 8 mm in size in transverse diameter. She underwent cystoscopy which revealed a solid 10 cm × 10 cm pedunculated lesion arising from the right lateral and anterior wall of the bladder, which was a rounded, mobile, well-circumscribed tumour with a smooth surface. The mucosa over the mass lesion and the adjoining bladder surface appeared intact. The right ureteric orifice was not seen and the left ureteric orifice looked normal. The tumour was bimanually palpable and mobile. She underwent transurethral resection of the tumour and histological examination of the specimen was reported to have shown nested variant of transitional cell carcinoma with marked atypical epithelial proliferation. Microscopic examination of the tumour revealed that the entire tumour was infiltrated by nests of polygonal cells with oval vesicular to hyperchromatic nuclei and eosinophilic to clear cytoplasm. Cystically dilated cells were also seen. The intervening stroma consisted of spindle cells with fusiform nuclei, compressing the cell nests in some areas, forming broad polypoid projections. There were areas of necrosis with acute inflammatory reaction. The metaplastic stromal cells exhibited no increase in mitotic activity. Krishnamoorthy and associates reported this case at a stage when there was no follow-up information regarding the outcome of the patient. Krishnamoorthy and associates [] stated the following.Nested variant of urothelial carcinoma can easily be confused with a number of benign lesions; it is very important for the pathologist to consider nested variant of urothelial carcinoma in the differential diagnosis of the lesions that show nested type growth pattern in lesions of the urinary bladder. It is equally important for the treating physician to adopt an aggressive approach towards the management of these lesions. The optimal treatment for nested variant of urothelial carcinoma is yet to be determined and this may be because of the rarity of the tumour, very small number of long-term survivors, and the absence of any randomized studies. The aggressive invasive growth and early metastases are the factors that favour radical cystectomy with adjunctive systemic chemotherapy. Nevertheless, a consensus is yet to be arrived at. They had reported their case in view of its rarity, its unusual histology, and its prognostic significance emphasizing the need to distinguish it from the classic transitional cell carcinoma. The aggressive behaviour of these nested variants underlines the importance of distinguishing them from benign proliferative lesions. Cytologic atypia is not a very good parameter because the mild atypia seen in nested variant of transitional cell carcinoma can be very deceptive, especially at low and medium power magnifications. Though the obvious invasion of the muscularis propria excludes the possibility of a benign lesion, the absence of invasion leads the pathologist onto a diagnostic dilemma.\nOoi and associates [] reported a rare presentation of nested variant of transitional cell carcinoma in a 74-year-old man who had bilateral hydronephrosis and acute renal failure. At cystoscopy, both ureters were obstructed with the right ureter narrowed along the entire length. Subsequent histopathologic examination from the ureteral resection revealed nested variant of urothelial carcinoma. Bilateral stents were inserted and the patient survived 12 months with a good partial response to chemotherapy. They stated that at the time of the report of their case in 2006, 76 cases of nested variant of urothelial carcinoma had been reported in the literature and at that time their patient was the first, to their knowledge, to present with bilateral hydronephrosis and tumour extension along one ureter. He had extensive liver and bony metastases and he eventually died 12 months pursuant to the establishment of the diagnosis. Ooi and associates [] stated that even though anecdotal reports of adjunctive chemotherapy with gemcitabine and carboplatin are being done, there are no available randomized studies on the effects of adjunctive chemotherapy in these patients with nested variant of urothelial carcinoma, after cystectomy. Furthermore, Holmäng and Johansson [] reported that there was no survival advantage with adjunctive radiotherapy in their series of seven patients with T stage in cystectomy specimens ranging from T1 to T4B.\nHolmäng and Johansson [] stipulated that nested variant of transitional cell carcinoma is aggressive and invasive, with a very well-differentiated histology, which is difficult to understand. Nevertheless, it had been postulated that the unusual histology may be due to the peculiarities of the host response mechanisms to carcinogenic stimulus such that the host is able to channel differentiation but cannot control invasion.\nTatsura and associates [] stated that the nested variant of transitional cell carcinoma has the characteristics of a focus of nests of transitional epithelial cells which infiltrate the lamina propria with apparent involvement of bladder mucosa. They also suggested that immunohistochemical analysis may help in the diagnosis of nested variants of transitional cell carcinoma derived from epithelial cells and that diagnosis and treatment at an early stage should reduce the mortality of patients with nested variant of transitional cell carcinoma.\nMurphy and Deana [] stated that the tumour cells of nested variant of transitional cell carcinoma are organized in nested structures and that many tumour cells are only slightly atypical, but a careful examination revealed that at least some significantly anaplastic cells are identifiable in each case, and the degree of anaplasia has a tendency to parallel the depth of invasion. They additionally stated that the features that identify this lesion as malignant are the tendency for increasing cellular anaplasia in the deeper portions of the lesion, its infiltrative nature, and the presence of muscle invasion. Mai and associates [] stated that despite the presence of mild or minimal cytological atypia in nested variant of transitional cell carcinoma, these neoplasms are occasionally associated with an aggressive clinical course and even death.\nDrew and associates [] reviewed the clinicopathologic features of 16 nested variants of transitional cell carcinoma over a 13-year period. They reported the following.Nested variant of transitional cell carcinoma was characterized by the presence of irregular nests and/or tubules of transitional cells infiltrating the lamina propria without surface involvement. The neoplastic cells tended to have innocuous features but at least a few cells in every case are cytologically anaplastic. There was a marked male predominance. Synchronous or metachronous transitional cell carcinomas of more usual histologic make-up may occur. After a follow-up averaging 16.6 months, only three patients were known to be alive with no evidence of disease.\nDrew and associates [] in 1996 made the ensuing concluding iteration.\nClinicopathologic information from their 16 cases combined with the 8 cases of nested variant of transitional cell carcinoma that were reported before the publication of their paper confirms that nested variant of transitional cell carcinoma is a persistent and aggressive neoplasm that is notable for its innocuous appearance in histologic preparations.\nLiedberg and associates [] reported three cases of the nested variant of urothelial carcinoma that were treated in their institution. They compared their outcome data with those of previously reported cases. They reported that the three patients presented with advanced muscle-invasive nested variant of urothelial carcinoma, of which two had lymph node metastasis at cystoprostatectomy. The histopathology in the latter two cases showed the same picture in the lymph node as in the primary tumour with nests of tumour cells with mild-to-moderate atypia. In all three cases the tumour involved the ureteric orifice or bladder neck. They concluded the following.Nested variant of urothelial carcinoma is a rare but an important histopathologic entity. Nested variant of urothelial carcinoma has a poor prognosis. At an early stage, the tumours might be difficult to differentiate from benign conditions and awareness of this condition is of outermost importance.\nBecause of the rarity of nested variant of urothelial carcinoma most urologists and pathologists would not have encountered a case of nested variant of urothelial carcinoma before and as a result of this there is the possibility that a case of nested variant of urothelial carcinoma may inadvertently be misdiagnosed. It is therefore pertinent to document iterations of Dhall et al. [], which summarize the microscopic features of nested variant of urothelial carcinoma as follows.These tumours are characterized histologically by large numbers of small, closely packed, poorly defined, confluent, and irregular nests of uniform urothelial cells infiltrating the lamina propria, reminiscent of von Brunn nests, and also infiltrating the muscularis propria with retained nested pattern (see Figures and ). These nests exhibit an infiltrative base as described by Volmar et al. []. Small tubules and microcysts may be seen as described by Talbert and Young [] and Young and Oliva []. The overlying urothelium may be normal in appearance. The cells comprising nested variant of urothelial carcinoma exhibit no significant cytologic atypia; they are mildly pleomorphic and show slightly increased nuclear-cytoplasmic ratio and occasionally prominent nucleoli (see ). Even though nested variant of urothelial carcinoma cells appears to be histologically bland, a number of authors [, , ] have observed significant pleomorphism, particularly within regions of muscle invasion. Mitotic figures are not readily seen. Mucin is not identified. The surrounding stroma varies from dense and collagenous to loose and myxoid or even oedematous. Lymphatic invasion may be seen []. In view of their deceptively bland appearance, the tumours are sometimes misdiagnosed as benign lesions, especially in the biopsy material leading in some instances to a significant delay in the establishment of diagnosis as stated by Young and Olive []. In some instances it is very difficult to establish an unequivocal diagnosis of nested variant of urothelial carcinoma in the biopsy material until multiple biopsies are performed. Nested variant of urothelial carcinoma must be differentiated from the benign proliferative lesions of the urothelium, such as von Brunn nests, cystitis cystica, cystitis glandularis, nephrogenic adenoma, paraganglioma, and inverted papilloma (see which illustrates von Brunn nests in comparison with nested variant of urothelial carcinoma showing regularly spaced urothelial nests with a relatively flat base).\nDhall and associates [] stated that the optimal treatment of nested variant of urothelial carcinoma is yet to be determined in view of the rarity of the tumour and in view of absence of randomized studies. They suggested that nested variant of urothelial carcinoma should be approached clinically as a high-grade disease with early cystectomy as an option for pT1 and pT2 tumours []. Dhall and associates [] additionally stated the following that.Adjuvant chemotherapy and radiation therapy have not been shown by a number of authors to be significantly beneficial in their reported series [, , ]. Nested variant of urothelial carcinoma should be kept in mind as a histologically unique variant which should not be confused with von Brunn's nest. Any bladder biopsy with tightly packed nests with any degree of architectural or cytological atypia should be evaluated with caution, and the possibility of nested variant of urothelial carcinoma should be raised in such circumstances.\nLinder and associates [] evaluated the oncological outcomes after radical cystectomy in patients with nested variant of urothelial carcinoma and compared survival to that in patients with pure urothelial carcinoma of the bladder. Linder and associates [] identified 52 patients with nested variant of urothelial carcinoma of the urinary bladder who were treated with radical cystectomy between 1980 and 2004. The pathological specimens were rereviewed by a single genitourinary pathologist. The patients were matched 1 : 2 by age, gender, ECOG (Eastern COOperative Oncology Group) performance status, pathological tumour stage, and nodal status to patients with pure urothelial carcinoma. Survival was estimated using the Kaplan-Meier method and compared with the log rank test. Linder and associates [] reported that the patients with nested variant of urothelial carcinoma of the urinary bladder had a median age of 69.5 years (IQR 62, 74) and a median postoperative follow-up of 10.8 years (IQR 9.3, 11.2). They also reported that nested variant cancer was associated with a high rate of adverse pathological features since 36 patients (69%) had pT3-pT4 disease and 10 (19%) had nodal invasion. Eight patients (15%) with nested variant cancer received preoperative chemotherapy. When the patients with the nested variant were matched to a cohort with pure urothelial carcinoma, no significant differences were noted in 10-year local recurrence-free survival (83% versus 80%, P = 0.46) or 10-year cancer specific survival (41% versus 46%, P = 0.75). Linder and associates [] concluded that the nested variant of urothelial carcinoma is associated with a high rate of locally advanced disease at radical cystectomy. However, when stage matched to patients with pure urothelial carcinoma, patients with the nested variant did not have an increased rate of recurrence or adverse survival. Linder and associates [] iterated that further studies are required to validate these findings and guide the optimal multimodal treatment approach to these patients.\nTripodi et al. [] reported the case of a 49-year-old woman affected by hepatitis C virus who presented with fever, discomfort, urgency, and hypertension. She had a computed tomography scan which showed a sclerosing inflammatory process that involved the connective and adipose tissue of the renal sinus. In absence of renal or pelvic masses felt that an underlying malignancy was excluded and renal abscess or tuberculosis was suspected. Accordingly, nephrectomy and proximal ureterectomy was performed. Tripodi et al. [] reported the following.Grossly, the calices, renal pelvis, and pelviureteric junction appeared modestly dilated with whitish, thickened, and uneven mucosa. Microscopic examination revealed that the subepithelial connective tissue, the fibromuscular layer, and the renal sinus fat were diffusely infiltrated by small nests of medium to large urothelial cells which were immunohistochemically stained positively with p63 and they had abundant eosinophilic cytoplasm and slightly atypical nuclei.\nTripodi et al. [] stated the following.On the basis of morphologic and immunohistochemical features, a diagnosis of nested variant of urothelial carcinoma was made. After surgery, the patient recovered from hypertension. Pelvic and upper urothelial tract nested variant of urothelial carcinoma was uncommon, and to the best of their knowledge, their case was the second case of nested variant of urothelial carcinoma with renal pelvis involvement.\nCerda et al. [] submitted an abstract for a poster presentation at the 25th European Congress of Pathology in Lisbon, Portugal (August 31st, to September 4th, 2013). They reported the case of 2 patients with nested variant of urothelial carcinoma as follows. |
A ten-year-old girl was admitted to our general hospital with numbness of her left palm and fingers in the last 5 months before admission. At that time, she was hit by a car while she was riding a bicycle. The car was coming from opposite side, and she fell with her left forearm was sliced by licensed plate of the car. There was a semicircular open wound with active bleeding on the left forearm, and she was in pain. She was brought to a nearby clinic and had her left forearm sutured. After the pain subsided, she felt numbness of her left hand and fingers. In addition, she could not extend her fingers. Finally, the patient decided to seek medical attention and get further treatment at our general hospital.\nFrom physical examination, there were claw hand deformity with thenar and hypothenar atrophy as well as a scar on the anterior side of distal forearm (). Sensorium loss of the palm and third, fourth, and fifth fingers was impaired. No tenderness was found. Capillary refill of the fingers was normal. Range of motion of the fingers was altered with limitation of finger abduction and thumb apposition (). Moreover, range of motion of the wrist was within normal limit.\nRoutine laboratory examination was within normal limit. The patient was taken for wrist and forearm radiographs and, similarly, there was no abnormality depicted on either bones or soft tissue.\nThe patient also underwent electromyography examination which showed median and ulnar nerve lesion at the left forearm with total axonal degeneration. No signs of reinnervation of both peripheral nerves were detected.\nThe patient was diagnosed as ulnar and median nerve palsy of left forearm, and then we planned to perform surgical exploration of the nerves and to repair with sural nerve graft, Zancolli procedure and sural nerve graft.\nIntraoperatively, skin incision was made on the previous surgical scar. Injury site was explored, and complete rupture of both ulnar and median nerves was found. Degeneration of both nerves was also seen, with neuroma rising from both the proximal stumps. The proximal and distal ends of both ulnar and median nerves was cut until nerve fascicle was visible. The distance between proximal and distal stump was measured: for ulnar nerve the distance was 7 cm, while it was 8 cm for median nerve. Sixteen centimeters of ipsilateral sural nerve was harvested, and the ulnar and median nerves were repaired using the nerve graft. Then Zancolli procedure was performed: skin incision was made along the palmar crease, A1 pulley was identified around metacarpophalangeal joint, longitudinal incision was made on the pulley, flexor digitorum superficial tendon was retracted laterally, metacarpophalangeal joint capsule was identified, an elliptical incision was made over the joint capsule, and capsulodesis was performed. Postoperatively the wound was closed and immobilized by elastic bandage ().\nWe followed the patient at 3-week postoperatively, and the patient had improvement of her claw hand (). She was advised to continue her rehabilitation of her hand to further improve her hand function, especially opposition and key pinch. At 6-month follow-up, she had improved grip strength and normal functional level of her left hand. At 2-year follow-up, she could handle daily activity as before the accident and was satisfactory with her condition. () |
A 28-yr-old woman presented with pain in both hips. The patient, a previously healthy housewife, had delivered her first baby about 3 weeks previously. Pain had first developed in the left hip some 3 months previously, at gestation week 32, and in the right hip 3 weeks previously (immediately after delivery). She had to use crutches because of disabling hip pain on weight bearing. At the time of presentation (postpartum 3 weeks, 3 months after left hip pain onset), the left hip pain was improving, but the right hip pain was worsening. Plain radiographs taken at 1 week postpartum showed diffuse osteopenia in both proximal femora, which was more marked in the left side (). Magnetic resonance (MR) images taken at 2 weeks postpartum showed a diffuse bone marrow edema pattern in the femoral head, neck and intertrochanteric areas of both femurs. No definite MR crescent sign () was detected (). The patient was treated conservatively under a diagnosis of TOH. Symptoms improved gradually and disappeared completely at 7 weeks postpartum (4 months after left hip pain onset) in the left hip and at 11 weeks postpartum in the right hip. Bone densities of proximal femora recovered in parallel with symptomatic improvement (). Follow-up MR images taken at 7 weeks postpartum showed markedly reduced bone marrow edema in the left and increased bone marrow edema in the right. ().\nCoronal multiplanar reformation (MPR) CT images were taken serially to evaluate the condition of trabecular bone of the femoral heads (). The first CT images taken at 4 weeks postpartum (4 weeks after right hip pain onset, and 12 weeks after left hip pain onset) showed marked decreases in the sclerotic densities of primary compression trabeculae with irregular discontinuation, more so in the right. In addition, a focal area showing irregular thickening of trabeculae was observed in the left femoral head, and in the right femoral head, breakages of subchondral bone with a linear dense sclerotic line crossing vertical trabeculae (suggestive a fracture line) were observed. On the second CT images taken at 11 weeks postpartum (11 weeks after right hip pain onset, 19 weeks after left hip pain onset), sclerotic densities of primary compression trabeculae had recovered markedly in both femoral heads. A focal area showing irregular thickening of trabeculae was also observed in the right femoral head. Third CT images taken at 20 weeks postpartum (20 weeks after right hip pain onset, 28 weeks after left hip pain onset) demonstrated increased trabecular density in both femoral heads, and the focal area of irregular trabecular thickening disappeared in both heads. The fourth set of CT images taken at 1 yr postpartum showed almost complete recovery of primary compression trabeculae in both femoral heads. |
AB is a 3-year-old-boy who came to the health centre because of different burns on his head and upper limbs. As the initial protocol of data collection, information was sent to the group, with photos of the child and the main problems. According to our data analysis, it was included in route 3. Our group concluded by the photos that almost 28% of the body had first- and second-grade burns caused by boiled water (head and left upper limb), which had subsequently scarred over. There was a persistent rigidness in the hand, with closed fist; in the face, his left eyelid could not be closed, and his mouth was not able to open, close, or move. He spoke by a little lateral gap between the lips and could not fed or maintain properly the food in his mouth.\nThe urgency of the case made the group give indications to the family for scar treatment while recovering more information about the adherence of the scars, functionality of the hand, and pain of the skin. According to the information returned, because of the eyelid damage, there was reduced visibility and an excessive dryness of the eye, which could lead to ulcers. We trained the personnel in Kalana in the Galveston technique, Kinesio Tape, and cohesive bandage through a specific protocol, and she gave the appropriate indications to the family about the use of both cohesive bandage and k-tape (the health centre could buy them to provide it to the family) to treat the adherence of the scars and eyelid, as well as the indications regarding cleaning and hydration of the scar to avoid limitations in joint range and mobility. Once a month, or once every two months, the family returned to the health centre so we could provide new indications about improving the mobility of the hand and the feeding.\nAfter one year of intervention, the child can now feed properly, holding the food within the mouth with a good use of the orofacial musculature without losing it. He can close his eye voluntarily and is able to do grasp and precision grips to participate in his daily living activities with the rest of the family. With this, he can join his family life. |
A 45-year-old male patient was referred to our pain clinic during admission for abdominal pain control due to chronic pancreatitis. He had been diagnosed with diabetes mellitus type 2, liver cirrhosis and hepatocellular carcinoma due to chronic alcoholism 5 years ago and was being followed up without any viable tumor recurrence after having received transcatheter arterial chemoembolization. His daily medications were 2 mg of glimepiride for diabetes, 10 mg of zolpidem tartrate and 75 mg of trazodone for depression and anxiety, and 10 mg of alfuzosin for benign prostatic hyperplasia. He had made frequent visits to the emergency room complaining of severe abdominal pain, and was admitted to the department of internal medicine for pain control and conservative management.\nThe patient's chief complaint was continuous epigastric pain and severe postprandial pain of 7/10 on a visual analogue scale (VAS) score of 0 to 10, which was dominant on the left side. The pain itself was well controlled at a VAS of 2/10 with opioid analgesics, but the patient experienced severe constipation and sedation and requested a nerve block in an attempt to reduce his pain medication. He was taking 40 mg of oxycodone four times a day and 8 mg of hydromorphone daily. The patient's most recent abdomino-pelvic CT showed no evidence of tumor recurrence, and atrophic change of the pancreas. Otherwise, there were no anatomical distortions within the patient's abdominal cavity.\nAfter obtaining a written consent form from the patient, we performed an elective left sided unilateral celiac plexus block under fluoroscopy guidance with the patient in the right lateral decubitus position. The patient's pain was clearly dominant on the left side, and because the source of his pain was a benign disease, a unilateral block was chosen in an attempt to reduce any possible complications. A skin wheal was raised with 1% lidocaine at a point 7 cm lateral to the L1 spinous process which corresponded to the inferior edge of the 12th rib, and a 15-cm 22-gauge needle was introduced at a 45 degree angle until it made bony contact with the L1 vertebral body. The needle's angle was adjusted as it was advanced by sliding off the anterolateral side of the vertebral body until it reached the anterior margin of the L1 vertebral body in the lateral view. A 10 ml plastic syringe filled with saline was then attached to the needle hub, and the needle was advanced slowly until loss of resistance was felt and pulsations of the aortic wall could be identified along the shaft of the needle. After the needle was aspirated and found to be negative for blood, a diagnostic block was done with 5 ml of 2% lidocaine mixed in 5 ml of iohexol (Omnipaque™, GE Healthcare Ireland, Carrigtohill, County Cork, Ireland) and adequate spread of dye was confirmed in the anterior-posterior and lateral view radiographs (). The patient was examined for any sensory or motor deficits for 20 minutes, and fluoroscopic evaluation was done to check for any delayed dye spread in unwanted regions such as across the midline of the vertebral bodies in the anterior view and posterior spread or along the psoas muscles in the lateral view. We checked for any numbness or dysesthesia in the T10-L2 dermatomes and weakness in quadriceps function as well as the patient's ability to wiggle his toes. When the patient's abdominal pain had resolved and no other side effects were observed, NCPB was done with 8 ml of 99% anhydrous alcohol (Dehydrated Alcohol Injection, Daihan, Ansan, Gyeonggido, Korea). The patient was followed up for 1 week until discharge from the hospital, and showed no motor or sensory deficits, hypotension or diarrhea. His pain medication was reduced to half of the pre-procedure dose, and the patient's pain was well controlled at a VAS score of 2/10. When the patient came to our out-patient clinic 1 month after discharge, his pain was still well controlled with reduced oral analgesics, but he reported that he had been unable to ejaculate since the procedure. He described his symptom as "a faucet turned off tight", and that he could not ejaculate during climax, which was a symptom that he had not experienced before the NCPB. We referred him to the department of urology and he is being followed-up under the impression of retrograde ejaculation but without any further evaluation. As of now, his ejaculation has not returned during the 3 months period after the procedure. |
Patient 4, a 15-year-old girl, was the only daughter of nonconsanguineous parents. Both her parents were healthy with no cutaneous anomalies. The girl was born at 40 weeks by normal delivery, with a birthweight of 3300 g, a height of 50 cm, and a head circumference of 35 cm. She first came to our observation at the age of 2 years, due to psychomotor delay. The parents noticed the presence of cutaneous spots at about 3 months of age. Since her first year of life, the girl showed mild body asymmetry. At physical examination, at age 2 years, the girl showed typical hypopigmented lesions along the lines of Blaschko, mainly in the trunk and the upper and lower limbs (particularly evident over the right part of the body). During these years, the girl manifested moderate cognitive delay, poor school performance, and generalized tonic-clonic seizures, with a frequency of 6 to 8 episodes per year despite valproate treatment. At the age of 6 years, clobazam was added to the valproate regimen and the frequency of seizures decreased. An EEG revealed generalized spike and wave discharges. Currently, her physical examination reveals a weight of 70 kg and a height of 174 cm (both falling in the 90th percentile). Her school performance remained poor. The diameter of the biceps region of the right limb was 3 cm larger than the contralateral. Her right thigh diameter was 66 cm and her left thigh diameter was 57 cm (Figure ): the sural right lower limb diameter was 43 cm, compared with the left, which measured 40 cm. The right lower limb length was 103 cm, whereas the left was 100 cm. Facial asymmetry was also present, but to a milder degree. She currently walks with unstable equilibrium with frequent falls down. A color Doppler ultrasound examination of her limbs showed normal laminar flow, with a wider diameter of the arteries in the affected right limb (Figure A and B). Brain MRI was normal as her ophthalmological examination. Spinal x-ray examination revealed mild left kyphoscoliosis. Skin biopsy was not performed. |
An 81-year-old, 77 kg, 183 cm man with a three-year history of muscle biopsy-proven IBM suffered a left intertrochanteric femoral fracture and a small parafalcine subdural hematoma as a result of falling down stairs in his home. The patient originally had progressive left proximal lower extremity and bilateral distal upper extremity weakness (left > right) that began several years before the diagnosis was definitively established with a muscle biopsy. The patient's serum creatine phosphokinase concentration was 748 U/L (normal < 170 U/L) at the time of the diagnosis. The patient had several near falls in the months preceding the accident. His left hand and wrist weakness affected his activities of daily living to some degree (fine motor skills), but physical and occupational therapy helped the patient remain active and he continued to live independently with occasional assistance from his wife. Because of worsening left quadriceps weakness, he had recently begun using a cane to help him rise from a seated position. The patient had a history of oropharyngeal dysphagia (solid food, pills) for which he was receiving care from an otolaryngologist. The patient also had chronic gastroesophageal reflux disease that was well controlled with a proton pump inhibitor. He did not have difficulty swallowing liquids and denied respiratory complaints associated with chronic aspiration. The patient did not lose consciousness after striking his head on the stairs and had no new neurological complaints. His other past medical history was notable for coronary artery disease, hyperlipidemia, chronic low back pain, and vertebral artery stenosis. He underwent coronary artery bypass graft surgery 16 years before the current admission. A previous transthoracic echocardiography revealed normal left ventricular systolic function. He denied chest pain, dyspnea, and exercise intolerance. The physical examination and radiographs of his femur revealed the left intertrochanteric fracture. The Glasgow coma scale was 15 and there were no new focal neurological deficits. A magnetic resonance imaging study demonstrated the small parafalcine subdural hematoma without a cerebral mass effect.\nThe patient was transported to the operating room for repair of the left intertrochanteric fracture. Because primary respiratory failure (, ) resulting, at least in part, from diaphragmatic dysfunction () has been previously reported in patients with IBM and prolonged duration of action of neuromuscular blockers may also occur, the authors opted to avoid the use of a neuromuscular blocker for endotracheal intubation and during maintenance of anesthesia in the current patient. After applying cricoid pressure, anesthesia was induced using intravenous lidocaine (0.5 mg.kg-1), propofol (1 mg.kg-1), and remifentanil (infusion of 0.1 mcg.kg-1.min-1). Manual positive-pressure ventilation with sevoflurane (inspired concentrations between 2% and 3%) in oxygen (100%) was used with sustained cricoid pressure to supplement to intravenous anesthetics after loss of consciousness. After uneventful endotracheal intubation, anesthesia was maintained with sevoflurane (end-tidal concentrations between 0.7% and 1.5%) and remifentanil (infusion rates of 0.05 to 0.15 mcg.kg-1.min-1). The orthopedic surgeon repaired the fracture with a trochanteric femoral nail. Intravenous acetaminophen and fentanyl were administered for postoperative analgesia. The patient tolerated the procedure well. He demonstrated vigorous spontaneous respiratory effort as he emerged from anesthesia and was extubated without difficulty in the operating room. The patient made an uncomplicated recovery from surgery and was subsequently transferred to a rehabilitation facility in stable condition. |
A 53-year-old diabetic man presented to our emergency department with history of the recurrent right loin pain in 2003. After evaluation he was diagnosed with right renal pelvis stone for which he underwent percutaneous nephrolithotomy (PCNL). The procedure was uneventful and the patient was discharged on the third postoperative day. A week after the PCNL, the patient returned to our emergency department with severe hematuria and clot retention. Routine blood investigations and coagulation parameters were within the reference ranges preoperatively. The patient was resuscitated with intravenous fluids and 3 units of packed red blood cells was transfused. His haemoglobin was 5 g/dL but coagulogram, serum electrolytes, and liver function tests and renal function tests were within normal limits. He had 2 additional episodes of severe hematuria with clots, despite conservative management. The urine was clear between the hematuria episodes. Ultrasound and computed tomography scans showed clots in the urinary tract without significant collection in the retroperitoneal space. The multiple episodes of gross hematuria which occurred during the period of conservative management changed the hemodynamic parameters, and the hemoglobin level decreased to 4 mg/dL; the patient underwent angiography which showed a pseudoaneurysm arising from segmental branch of right renal artery, although there was no active contrast leak. Later angioembolization was done with a metallic steel coil in 2003. Following the procedure his hematuria was settled and he was discharged in the satisfactory condition.\nHe remained asymptomatic till 2011 when he started developing bilateral dull aching loin pain. He presented to our outpatient department in 2012 with the increasing loin pain more on right side than left. He was evaluated and X-ray KUB was done which revealed a 2.7 cms renal pelvic calculus with the embolization coil in its center with multiple calyceal stones in the right pelvis and a left upper ureteric calculus (). His routine haemogram, coagulogram, serum electrolytes, and liver function tests were within normal limits. His serum creatinine and urea were 1.0 mg/dL and 27 mg/dL. Vitamin D3 and PTH (parathyroid hormone) were within normal limit, that is, 52 nmol/L and 21 pgm/mL, respectively, and urine culture was sterile. His random blood sugar was persistently high in the range of 240–290 mg/dL, so he was switched from oral metformin to injectable regular insulin on sliding scale in view of scheduled surgical candidate.\nHe was taken for surgery and he underwent left laparoscopic ureterolithotomy and right percutaneous nephrolithotomy (PCNL). Intraoperatively the migrated stainless steel embolization coil was seen being engulfed all around by the multiple stones in right pelvis (Figures and ). We used pneumatic lithotripsy and complete retrieval of the all pelvic calculi along with the migrated embolization coil was done (). Postoperative period was uneventful. He did not have any significant bleeding and his blood sugar was maintained within normal range. Nephrostomy tube was removed on the postoperative day 4 and following which he was discharged on the postoperative day 5 on injectable insulin glargine and insulin lispro. Later he was followed in outpatient department and was doing well. |
A 55-year-old man with a history of atrial fibrillation and smoking but without a family history of stroke was admitted to our hospital with a 5-hour history of left limb weakness and speech difficulties. The heart rhythm was uneven, and the first heart tone was neither strong nor weak. He had mixed aphasia, eye gaze to the right, shallow left nasal and labial groove, mouth angle slanted to the right, left limb muscle strength grade 1, positive left side pathological signs, and National Institutes of Health Stroke Scale (NIHSS) score of 16 points. Emergency brain computed tomography (CT) showed a large cerebral infarction in the right frontal temporal parietal lobe. Chest CT revealed pulmonary edema and double lung inflammation.\nThere was a need to recanalize because of cerebrovascular occlusion, and the family members of the patient agreed to undergo endovascular interventional therapy. After successful right femoral artery puncture, the angiography guidewire could not pass through the aortic arch (Fig. A). 5F pigtail angiography showed that there was no development of the aortic arch or vessels above the arch. Considering the vascular variation, we performed a right radial artery puncture. Cerebral angiography was performed again and aortic arch interruption was clearly diagnosed (Fig. B). The right internal carotid artery was occluded (Fig. C), and the posterior communicating artery was opened. Considering the possibility of cardiogenic embolism, a middle catheter was used for thrombus aspiration of the right internal carotid artery, and a dark red thrombus was removed. Angiography showed that the right middle cerebral artery, right anterior cerebral artery trunk and distal branches were clearly visible (Fig. D), no luminal stenosis was found, modified thrombolysis in cerebral infarction score forward blood flow was grade 3, and the operation was successful.\nNo hemorrhage was found on brain CT immediately after the operation and on postoperative day 1. Treatment to improve circulation, anticoagulation, control ventricular rate and anti-infection were administered. Unfortunately, oxygen saturation decreased on postoperative day 3, and he underwent tracheal intubation. The NIHSS score decreased to 4 on postoperative day 6. The patient was breathing smoothly on postoperative day 7, and tracheal intubation was successfully performed. Magnetic resonance imaging showed a large area of right-side acute cerebral infarction and small hemorrhage after infarction in the right basal ganglia, and the right internal carotid artery was unobstructed (Fig. A, B). Computed tomography angiography indicated that the aortic arch was interrupted (A type, congenital cardiovascular malformation), and multiple collateral vessels were found (Fig. A, B). The NIHSS score decreased to 1 point at 1 month after the operation, which satisfied the patient and his family members, and the patient was discharged after improvement of his condition. The patient and his family refused further treatment for aortic arch interruption. The modified Rankin Scale score was 0 at 3 months and 1 year postoperatively, indicating a good prognosis. |
A 48-year-old male with a past medical history of cystic acne, but otherwise insignificant medication and social history, presented with concern for a parasite on his face. He reported that on the previous night he thought a parasite had crawled out of a healed cystic lesion on his right cheek. He presented a jar with a bloody napkin and paper, stating that he had scraped the parasite from his face with a scalpel and brought it with him to the ED. His physical exam was notable for diffuse excoriations and scaly patches, a 3 × 4 centimeter (cm) abrasion on his right cheek and mild anxiety. Otherwise his vital signs and physical exam were unremarkable.\nNo obvious parasites were appreciated on examination of his skin or in the jar. Basic blood tests including a complete blood count (CBC) and basic metabolic panel were within normal limits. A urine toxicology test was evaluated and reported negative. During his stay, the patient called a provider to the bedside to evaluate his thumb for a parasite actively crawling out of his skin. The provider documented that no obvious insects or wounds were apparent at that time. Delusional parasitosis was suspected and psychiatry was consulted, but the patient eloped from the ED prior to being evaluated.\nThe patient returned to the ED two days later with a complaint of parasitic infection. This time he described seeing white maggots crawling from his skin and presented us with a jar with a flaky material. The patient presented to the outpatient infectious disease (ID) clinic and was directed to come to the ED to obtain a formal ID consult. Although there was low clinical suspicion for an infectious component, ID was consulted for the presumed benefit of reassurance from a consultation service. The flakes presented by the patient were analyzed for ova and parasites and reported to the patient as negative. He was then evaluated by psychiatry who reported that his presentation was consistent with a delusional parasitosis but did not recommend inpatient hospitalization given that he did not appear to be at risk for harm to himself or others. He eloped a second time prior to receiving discharge papers.\nThe patient returned for a third visit to the ED with similar complaints. His workup included basic labs and a non-contrast head computed tomography (CT), which were all unremarkable. He was instructed to follow up with psychiatry as an outpatient. |
An 81-year old female with a history of coronary artery disease, hypertension, and thrombocytosis suffered a witnessed trip and fall onto a nightstand. The patient took 75 mg of clopidogrel daily in addition to an 81 mg aspirin tablet. She reported a mild headache however had no change from her baseline mentation per family members with no evidence of obvious injury aside from a small area of ecchymosis near a small forehead laceration. She remained up and ambulatory with no further complaints. Ten hours after her injury the patient presented to the Emergency Department with stridorous and agonal respirations with a profoundly decreased level of consciousness. She was noted to have developed extensive ecchymosis on the anterior portion of her neck and chest. Her symptoms had begun rapidly shortly prior to arrival while lying in bed. Family reported that she had been in the constant company of her husband with no further falls or injuries that had occurred since her fall. The patient was intubated upon hospital arrival due to respiratory extremis with obvious swelling and crepitus noted on neck examination. A noncontrast CT scan of head was unremarkable while there was demonstration of a large retropharyngeal hematoma measuring 3.6 cm by 5.3 cm by 20 cm on a CT of the cervical spine with no evidence of fracture. Her hemoglobin was 9.5 gm/dL and platelets were 1234 per deciliter, with an INR of 3 and a slightly below normal and activated partial thromboplastin time of 23.9 seconds (reference range 25-35 seconds). A CT angiogram of the neck was subsequently obtained demonstrating active bleeding from the anterior ligaments of the vertebral column that was not felt to be amenable to embolization (). Given the extent of the hematoma intraoral surgical evacuation was performed with bleeding from the anterior vertebral spine controlled with Bovie cauterization, placement of topical thrombin, and drain placement. No reaccumulation of hematoma was noted during her hospital course. The patient unfortunately expired 12 days from the date of admission from presumed aspiration pneumonia and multisystem organ failure. |
We present the case of a 77-year-old ambulatory man with hypertension, sarcoidosis, complete atrioventricular block status post-pacemaker implantation, chronic kidney disease due to FSGS, and right facial nerve paralysis, who presented with sporadic gait and right face numbness. He was diagnosed with sarcoidosis by biopsy of a tumor in front of the right tibia 14 years before presentation. Since the tumor and abdominal lymphadenopathy were the only manifestation of sarcoidosis and no other signs of organ involvement were present, he received no immunosuppressive treatment. The abdominal lymphadenopathy had been stable over time. Nine years before presentation, he was referred to our nephrology clinic to determine the cause of chronic kidney disease. His serum creatinine level was 1.2 mg/dL and he had proteinuria of 0.4 g per day. Hematuria was not present. Renal biopsy revealed six globally sclerotic glomeruli among all 34 glomeruli (18%) and some residual glomeruli with segmental sclerosing lesions, but no involvement of sarcoidosis. He was diagnosed with primary FSGS. Since the proteinuria was mild, he did not receive immunosuppressive treatment.\nOne year after that, the patient experienced palpitations and was diagnosed with complete atrioventricular block. Coronary angiography showed no significant stenosis of the coronary arteries, and he underwent pacemaker implantation. Whether sarcoidosis contributed to the complete atrioventricular block was unclear. The abdominal lymphadenopathy and the dyskinesia of the ventricular septum were stable and did not progress over time.\nThe patient was stable for eight years, until when he started to suffer from sporadic gait and right face numbness that occurred and resolved within a day every few weeks. Three months later, the symptoms recurred along with sudden dysarthria and left limbs weakness. Physical findings were notable for pronator drift on the left side. Perfusion computed tomography (CT) with iodinated contrast and CT angiography revealed no ischemic lesions or occlusion of major cerebral arteries. The symptoms disappeared three hours after the onset. A transient ischemic attack (TIA) was suspected, and he was admitted to the stroke unit. Ultrasonography revealed no stenosis of the internal carotid arteries, and transesophageal echocardiogram showed no abnormalities of the atrial septum. His pacemaker detected paroxysmal atrial fibrillation, which was presumed to be the etiology of the TIA. Thus, edoxaban 30 mg per day was started and he was discharged after one week of hospitalization.\nOne month after his discharge, his left leg started to swell and his gait worsened. Urinary protein excretion was 0.6 g per day, serum creatinine was at the baseline level of 1.6 mg/dL, and serum albumin level was 3.8 g/dL. Although no coagulopathy was found, ultrasonography revealed left femoral vein thrombosis that was 41 mm long. Edoxaban was stopped, and heparin was administered intravenously for two weeks. Low mobility due to his gait was presumed to be the cause of development of deep vein thrombosis (DVT). The patient was switched to warfarin and was discharged, but the left leg edema persisted. Three months later, he developed complications of urinary retention and constipation.\nFour months after discharge, the patient presented to the emergency department with sudden left leg pain and inability to walk. The entire left lower limb was slightly pale and had slow pitting edema. The left dorsal artery was not palpable, and the left femoral artery was barely palpable. Contrast CT revealed occlusion of the left femoral and superficial femoral arteries together with the known DVT in the left femoral vein (Fig. , ). Emergency thrombectomy for acute arterial occlusion was performed and the leg perfusion resumed. The emboli (maximum of 23 mm in diameter) were sent for pathological examination. The patient was admitted to the hospital and started on heparin infusion in place of oral warfarin. The history of recent TIA implied hypercoagulable state, but again no coagulopathy was found. While malignancy screening was being planned, the pathology of the arterial emboli revealed an unusual and surprising finding: the surface of the thrombi was filled with large atypical lymphoid cells (Fig. ) and was covering the necrotic interior of the thrombi. Immunohistochemical analysis showed that the tumor cells on the surface and the necrotic interior of the thrombi were positive for CD20 and CD79a but negative for CD3 (Fig. , ), which is characteristic of B cells. Leukocytosis was absent (white blood cell, 4,000/μL; segmented neutrophil, 55%; lymphocyte, 34%; monocyte, 9%; eosinophil, 2%). Serum soluble interleukin-2 receptor level was 1,548 U/mL (normal, 122–496 U/mL); lactate dehydrogenase (LDH) level, 808 U/L (normal, 120–245 U/L); LDH-2 fraction, 39% (normal, 28–35%), and LDH-3 fraction, 32% (normal, 21–27%). These findings were consistent with large B-cell lymphoma with intravascular proliferation, but the etiology of the aortic thrombi was unclear.\nThe hematology consultation team considered that the patient needed further biopsy to determine the etiology. Bone marrow biopsy showed normocellular marrow with normal maturation, but with infiltration of CD79a-positive large atypical lymphoid cells within the small vessels (Fig. , ). Although no lymphadenopathy was detected on palpation, CT scan showed swollen bilateral axillary and inguinal lymph nodes, which were up to 30 mm in diameter. While surgical biopsy of the right axillary lymph node and random skin biopsy were planned for diagnosis, the patient developed a complication of sepsis presumably due to pyelonephritis on hospital day nine. Piperacillin/tazobactam and vancomycin were started. Because partial thromboplastin time was prolonged, biopsies were withheld. Although white blood cell and neutrophil counts were improving, the patient died due to sudden respiratory and cardiac arrest on hospital day twelve. The patient had a do-not-resuscitate order. His family agreed to an autopsy. |
A 45-year-old woman presented to our hospital with multiple lung nodules. She had a history of poorly differentiated thyroid carcinoma, diagnosed 7 months prior to admission, at an outside hospital. The patient was healthy otherwise and reported no radiation exposure or any family history of thyroid cancer. The initial work-up at the time of discovery of the right thyroid nodule included fine needle aspiration and core biopsy, with findings consistent with poorly differentiated thyroid carcinoma. The patient then underwent a total thyroidectomy and central neck lymph node dissection. The pathologic diagnosis from the outside hospital reported a 2.8 × 2.4 × 1.1 cm tumor in the right thyroid without extrathyroidal extension or lymph node metastasis. However, both capsular invasion and extensive vascular space invasion were noted. Based on the tumor size, tumor extension and lymph node status, the tumor was designated as Stage II (pT2 pN0 pMx). IHC staining showed that the tumor cells were positive for thyroglobulin and thyroid transcription factor 1 (TTF1). An immunostain for p53 was also performed at the outside hospital and showed a small focus (< 1 cm) with p53 positivity, suggesting a diagnosis of anaplastic thyroid carcinoma.\nAt our institution, the diagnosis was revised, based on review of both the primary thyroid tumor and the current lung metastases. Both tumors were remarkable for biphasic malignant components: the carcinoma and the sarcoma. The carcinoma component showed a poorly differentiated microfollicular type thyroid carcinoma, composed of sheets and islands of tightly packed thyroid follicles with dense colloid. The tumor nuclei were small and round with vesicular chromatin, resembling those of typical poorly differentiated follicular thyroid carcinoma. Admixed with the epithelial component were malignant spindle cells with small round blue cell type morphology. Focally, rhabdomyosarcoma-like cells with eosinophilic cytoplasm were appreciated. No heterologous cartilage or bone components were identified. The IHC staining performed at the outside hospital showed that the thyroid carcinoma (epithelial) component was positive for thyroglobulin, PAX8 and TTF1 (Fig. ). The sarcoma (spindled) component was negative for all thyroid carcinoma markers (TTF-1, thyroglobulin and PAX8), but was positive for vimentin and focally positive for myogenin (supporting skeletal muscle differentiation) consistent with mesenchymal differentiation. Interestingly, the foci of vascular space invasion contained both epithelial and mesenchymal components as well.\nThe patient received Taxol with Carboplatin for 7 weeks followed by radiation therapy. Her thyroglobulin level rose from 1.2 ng/mL to 25.40 ng/mL 5 months after completion of the chemo-radiation therapy, suggesting progression of the disease. A follow-up CT scan of the chest showed multiple newly developed nodules (ranging from 1 to 2 cm) in the right lung, highly suspicious for metastases. The patient underwent a right thoracotomy, right lung resection/metastasectomy. The surgery was uneventful with negative resection margins. However, the patient’s general condition deteriorated and she succumbed to the disease 4 months later.\nHistological examination of the lung nodules revealed similar tumor morphology and tumor differentiation when compared to the original thyroid tumor, which is somewhat unusual for a biphasic carcinosarcoma (Fig. ). Tumor necrosis was also present. Mutational analysis using a next-generation sequencing based assay showed that the neoplastic cells from the lung metastasis were devoid of genomic alterations for known thyroid cancers, including BRAF, RAS family (KRAS, NRAS and HRAS), EGFR, PTEN, TERT, PI3Kinase or RET. BRAF or RAS family are known as the most commonly altered genes in papillary thyroid cancers. Other molecular mutations reported in the development of anaplastic thyroid carcinoma include p53, PAX8/PPAR gamma rearrangement []. None of the mentioned gene mutations were identified in our patient.\nHowever, an interesting finding in this case is the presence of a point mutation in DICER1 (E1705K) that has previously been associated with differentiated thyroid carcinoma [, ]. Whether the DICER1 (E1705K) mutation is the underlying genetic event leading to the initiation of tumorigenesis or is downstream to other gene alterations in tumor development is largely unknown. Additional mutations of unknown significance were also detected in this tumor including FLCN (R239H), POLD1 (Q684H) and SYK (R217L). These variants have not been adequately characterized in the scientific literature and their prognostic and therapeutic significance is unclear. |
A 50-year-old female presented to the emergency department with complaints of weakness, right lumbar pain, and painless total hematuria. She had lost 20 kg of weight in the past 2 months and her hematuria had been present for the past month with no history of trauma. The physical examination was unremarkable except for increased sensitivity on her right flank and hemoglobin and hematocrit values of 6.9 mg/dl and 20%, respectively. Owing to clot retention, continuous bladder irrigation was initiated and her hemoglobin levels were corrected with transfusions. Abdominal ultrasonography revealed bilateral renal masses. Computed tomography (CT) scans of the chest, abdomen, and pelvis showed a 9 cm cystic renal mass on the right and another 5 cm tumor on the left kidney; a subpleural 5 cm lesion was visible on the right lower lobe of the lung CT (, ). Bone scans showed no evidence of metastasis. An ultrasound-guided tru-cut biopsy of the right kidney was performed with a pathologic diagnosis of xanthogranulomatous pyelonephritis. Fine-needle aspiration biopsy of the lung lesion was reported as the coagulation type of necrosis. After cystoscopic evaluation to disclose the source of the hematuria, right transperitoneal radical nephrectomy was performed. The surgery was uncomplicated except for a 3 cm tear in the vena cava that was primarily sutured.\nOn the first postoperative day an increase in serum bilirubin levels was detected and the patient developed tachypnea and dyspnea. A repeat CT scan of the chest and abdomen demonstrated multiple lesions in the liver and spleen that were not visible on the preoperative scans, with an increase in size and number of the lesions in the lung. The patient's recovery period was troublesome and extended; nevertheless, no further surgical intervention was necessary. The pathologic report of the removed kidney was pure choriocarcinoma (); assessment of the serum beta-human chorionic gonadotropin level was over 1 million mIU/ml. The final diagnosis was gestational choriocarcinoma, and the patient was referred to the oncology department for methotrexate-based chemotherapy following stabilization of her general status. |
A 69-year-old Filipino man with history significant for hypertension and hyperlipidemia presented to his primary care physician with hematuria with weight loss of 1 month’s duration. He did not have any flank pain, burning on urination, or increased urinary frequency. He did not endorse any symptoms of fatigue or night sweats. His only medication was atenolol for his hypertension. He did not smoke tobacco, drink alcohol, or do any recreational drugs. He was unemployed at time of interview. He did not have any family history of cancer. His vital signs were within normal limits. On physical examination, he was well appearing and in no acute distress. He had no palpable mass and had an otherwise normal cardiovascular, respiratory, and neurologic examination. Laboratory work showed normal cell counts and normal electrolytes; the results of his kidney and liver function tests were normal. A computed tomography (CT) – intravenous pyelogram was performed as a diagnostic work-up for his hematuria, which demonstrated a large mass in the left collecting system and proximal ureter. He was seen by urology with plans for surgical resection 1 month later. Three weeks later he was admitted to the Emergency Department with nausea and vomiting. He was tachycardic to 110 beats per minute but maintained a normal blood pressure. His laboratory results were notable for hemoglobin to 12.1. His sodium was 134. At that time, a CT scan of his abdomen and pelvis showed interval enlargement of the left renal mass. An ureteroscopy with biopsy was performed, which showed necrotic tissue with rare crushed degenerating atypical cells. A screening chest CT scan was also obtained which showed a small 3 mm nodule in the lower lobe of his left lung. A follow-up interventional radiology-guided left kidney biopsy showed a cellular neoplasm with sheets of pleomorphic round cells with hyperchromatic nuclei, irregular nuclear outlines, and inconspicuous nucleoli with scant and delicate cytoplasm which is consistent with SCC. The tumor cells were positive for the neuroendocrine markers synaptophysin and CD56 with focal staining for chromogranin and dot-like positive staining for cytokeratin (AE1/AE3), supporting the diagnosis of SCC (Fig. ). A bone scan did not show any metastatic lesions. Shortly afterwards, he developed dizziness and an MRI of his brain was obtained revealing a 1.6 cm partially hemorrhagic round mass with surrounding edema in the midline superior vermis potentially representing metastatic disease. An additional 4–5 mm hemorrhagic metastatic focus was seen in the right occipital convexity. The cerebellar mass was resected and probably represented a renal origin due to the absence of lung masses along with clinical and radiographic correlation. He was started on whole brain radiation therapy during his in-patient stay. An out-patient oncology referral was made but he was unable to establish care due to frequent hospitalizations. He had several hospital admissions for nausea and vomiting and continued to decline functionally. He developed chronic hyponatremia during these hospitalizations which were attributed to SIADH. He originally presented with sodium of 119 and was stabilized to a sodium level of 128 with the use of salt tablets. He declined chemotherapy when it was offered by the oncology team during in-patient consultation due to poor quality of life and functional status; he died within 8 months of presentation at his nursing facility. The cause of his death was unknown. An autopsy was not performed. |
34 years old patient, Gravida 6 Parity 3, previous 2 miscarriages (18 weeks & 12 weeks), was seen first at 23 weeks 4 days of pregnancy. She had undergone previous 3 cesarean sections and an evacuation of retained products of conception by curettage in 2013 for partial hydatidiform mole. At 27 weeks 5 days, she was admitted for vaginal bleeding. On further evaluation by ultrasound (), the diagnosis of placenta percreta was made (later confirmed by MRI). At 29 weeks, she had constipation with 2 episodes of urinary retention and she was put on continuous bladder drainage. She developed urinary tract infection and treated with appropriate antibiotics based on culture sensitivity. She continued to have repeated bouts of vaginal bleeding of varying amounts and severe constipation from 31 weeks of gestation.\nAt 32 weeks 4 days, patient underwent cystoscopy, which had shown signs of cystitis with no definite infiltration. She underwent classical cesarean section under combined anesthesia (Epidural + General). The umbilical cord was tied near insertion and the placenta was left in situ because there was no spontaneous separation. Then, the uterus was closed. Prophylactic temporary bilateral internal iliac artery balloons were inserted and inflated earlier. Uterine artery embolization was performed post cesarean section and selective angiograms confirmed adequate positioning. The patient required large volume of particles and still had incomplete embolization with the lower part of the uterus still showing some unblocked branches on both sides.\nPost-operatively, she was transferred to labor ward and within 4 hours, she developed clinical features of pulmonary embolism (PE). Some of her symptoms included drop in O2 saturation to 81%, tachycardia, chest pain, peripheral cyanosis, and signs of respiratory distress. Then, she was transferred to ICU and was initiated on heparin infusion. On chest X-ray, she had no atelectasis, pneumothorax, or pleural effusion. An immediate CT scan did not show any PE. There was no Doppler evidence of venous thrombosis in the femoral and popliteal venous systems. Later on day 1 post-operative, she had focal patchy consolidation left base and was started on parenteral meropenem, linezolid and fluconazole for the next 5 days. She had two consecutive CT scans on post-operative on days 2 and 3, which were negative. On ECG, there was right heart strain. She was now on enoxaparin. On the post-operative day 5, she was prescribed parenteral piperacillin-tazobactam for 5 days and she was shifted out of ICU next day. She had 500ml vaginal bleeding on the 9th post-operative day. 2 units PRBC were transfused. She was switched to oral cefuroxime and metronidazole and planned to continue on long-term low dose antibiotic. On post-operative day 11, she received methotrexate. On day 12, the MRA had shown the placenta was still enhancing with some areas of infarct and separation, fluid collection in the uterine cavity (present from day 1 post op, not increasing), with large ovarian veins, hugely distended and extensive pelvic varices, R>L, extensive collaterals. Her CRP was 12.7 mg/L.\nOn post-operative day 13, she underwent total abdominal hysterectomy. Intraoperatively, the bladder was densely adherent, drawn up, with large vessels in the broad ligament. The lower segment was bulging due to the presence of the placenta. The uterus was about 24 weeks’ size with adherent omentum. There was 100 mL of old blood in the cavity and the placenta was partially infarcted. The total blood loss was 2000 mL.\nPost-operatively, she was in ICU for 2 days receiving anticoagulation treatment (bridging treatment with enoxaparin + warfarin) and patient controlled analgesia. She had a bout of severe cough on day 4 and loose motions on day 5. She was diagnosed with vault hematoma, which was retro-vesical, about 120 ml in volume, treated conservatively. On day 10 she had been discharged from the hospital.\nShe presented to the ER on the post-operative day 16 and was diagnosed with chronic pulmonary embolism. Patient had a pulmonary embolus within the right middle lobe pulmonary artery; areas of sub-segmental embolus within the right lower lobe pulmonary arteries. She had no pleural effusions or consolidation and no mediastinal lymphadenopathy. She was readmitted for 4 days. She was started on therapeutic enoxaparin + warfarin. She was continued on 6 mg warfarin for 4 weeks after discharge. |
We present a case study of a 56-year-old community ambulating male who endured a failed MTP arthrodesis due to infection, whom we treated with a novel method of creating an articulating antibiotic impregnated cement spacer. The patient had undergone a right first MTP arthrodesis with fixation via three 4.0 mm cannulated screws by another provider one year and four months earlier. A month following the arthrodesis, the patient was found to have a purulent draining wound with cultures growing Methicillin-Resistant Staphylococccus Aureus (MRSA). During the course of the year after diagnosis, the patient was placed on suppressive antibiotics and he managed his wounds with appropriate means for the intermittent drainage. Postoperative radiographs after a year demonstrated a lack of definitive fusion and lucencies surrounding the screw implants indicative of osteomyelitis as seen in Figure .\nOn physical exam, his right first ray was swollen, erythematous, and warm without any open wound or sinus tracts. The patient was neurovascularly intact and an initial radiograph in the office revealed failed hardware. Due to his history and clinical presentation as well as the concern for potential seeding to his hip arthroplasty implants, recommendations to remove the foot hardware and eradicate the infection with placement of an antibiotic spacer were presented. After a thorough discussion of alternative measures, and risk and benefits, the patient agreed to move forward with the removal of hardware and placement of an antibiotic spacer. A preoperative presentation of the patient's foot is seen in Figure .\nThe patient was aware and agreeable to the case presentation, and consented during the time of the operative procedure.\nA dorsal midline incision was made incorporating the previous incision, and the first MTP capsule was then incised allowing visualization of the area of nonunion. After identifying the osteomyelitis around the hardware and the metatarsophalangeal joint, the two intact screws were removed under fluoroscopic guidance. The final screw fragments were removed with the trephine technique by flexing the interphalangeal (IP) joint and creating a core to pull out the proximal piece. Gross purulence was noted medially over the broken hardware, which was sent for culture and speciation along with the removed hardware.The MTP joint was then resected proximally and distally to remove all portions of diseased bone as seen in Figure .\nAn antibiotic spacer was created using 1 gram of vancomycin, 40 grams of bone cement, and a 10 ml syringe for molding purposes. The antibiotic impregnated cement was inserted into the syringe. Using the plunger within the syringe, a concave surface was created by the abutting rubber portion of the plunger. An antibiotic spacer stem/keel was fashioned through the cement that was pushed into the tip of the syringe. Once the cement had hardened, the plastic of the syringe including the narrow tip was removed using a micro-sagittal saw, providing an 8 mm cylinder of cement. The mold was resected to be approximately 8 mm in length to maintain the length and orientation of the joint, and the narrow tip provided a keel for insertion into the metatarsal as seen in Figure . The MTP joint was then thoroughly irrigated with 3 liters of antibiotic impregnated solution, and the spacer was then inserted (Figure ).\nThe joint was assessed for proper tensioning and stability through limited ranges of motion and stressing in all planes. The capsule was closed with 3-0 vicryl suture, and the skin was closed with 3-0 nylon suture. Xeroform was placed over the incision and a dry sterile dressing was applied with an overlying posterior mold with sugartong fiberglass splint. While in recovery, post operative radiographs were taken (Figure ).\nPostoperatively, the infectious disease team saw the patient and a peripherally inserted central catheter (PICC) line was placed for administration of intravenous vancomycin and ceftriaxone. On postoperative day number 1 a baseline erythrocyte sedimentation rate (ESR) was collected which measured 34 mm/hr (Normal 0-28 mm/hr) and serum white blood cell count (WBC) of 8,800/uL (Normal 4,500-10,000/ uL). Intraoperative cultures remained negative for bacteria and fungi. His clinical stay remained uneventful and he was discharged home on postoperative day number 4.\nHe presented to the office for follow-up one week postoperatively with a clean and dry wound without drainage or evidence of cellulitis. The PICC line was functioning well and the patient was placed in a hard soled shoe with eventual progression to a walking boot. On postoperative day 15, no signs of infection were noted and his sutures were removed after successful wound healing.\nOne month postoperatively, the patient presented to the ED with complaints of his PICC line bleeding. While being evaluated in the ED, the patient was found to have ipsilateral thromboses within the axillary and subclavian veins. The patient was placed on a blood thinning regimen, which included lovenox and a coumadin bridge. The patient was subsequently changed from intravenous ceftriaxone and vancomycin to oral doxycycline. The thrombosis was attributed to the underarm trauma from the use of crutches and the patient was provided education and physical therapy with the use of a walker. At his most recent follow-up, approximately one year postoperative, he denied any pain to his foot and was adamantly against having the cement implant removed. He continues to have an active lifestyle, which is free of any limitation. |
Our patient was a 56-year-old postmenopausal African American woman with no past medical history who was previously treated at an outside oncology clinic for breast masses until 2010, when we first saw her. Her family history was negative for breast or ovarian carcinoma. She had a negative smoking history and endorsed drinking one drink per week. Per reports obtained, she first presented in 2006 with left breast lesions located in the upper inner breast that were documented as complicated cystic masses within the 9 o’clock and 9:30 positions on the basis of ultrasound (US). Subsequent US core biopsy in both areas revealed intraductal papilloma (IDP), and the patient was referred for a surgical consultation. No additional documentation of clinical visits was available until 1 year later. That documentation was in the form of a core biopsy pathologic report documenting the patient’s history of IDP at 9:00 and 9:30 positions as well as intraductal papillomatosis of the breast. A core biopsy taken at that time was from the left breast (location not mentioned) as well as the left axilla. The patient’s left breast showed fibrosis of mammary stroma including intralobular stromal sclerosis as well as microcalcifications in the lobular lumens. An axillary core biopsy confirmed IDP of the breast with stromal hyalinization as well as lymph node tissue adjacent to the papilloma. One month later, she underwent lumpectomy, with pathology reporting a 7-mm intracystic papilloma within a lymph node that was completely excised, as well as an epidermal inclusion cyst. The pathologist noted that the tumor was located near the periphery of a lymph node, probably arising in ectopic breast tissue in the capsular region. Approximately 11 months later, she developed another left breast mass. This was excised after a US-guided core biopsy once again revealed a benign IDP. The patient was then lost to follow-up at the outside clinic. She presented to our clinic 2 years later for an evaluation of a new left breast lesion. A bilateral diagnostic mammogram revealed two masses in the left breast, which were not well visualized, owing to heterogeneously dense breast tissue. Diagnostic US revealed a solid superficial mass measuring 0.81 × 0.76 × 0.81 cm corresponding to palpable findings also seen at the 6 o’clock position (Fig. a). Additionally, the patient had a large, complex cystic mass measuring 7 cm at the 1 to 3 o’clock position abutting the pectoralis muscle (Fig. a). A core biopsy of the 6 o’clock lesion was recommended. A US-guided, vacuum-assisted core biopsy of the 6 o’clock mass revealed an intracystic papillary neoplasm. Per the report, the patient denied nipple discharge, dimpling, thickening, redness of the skin, swelling, or tenderness at the time. A few weeks later, she underwent left breast lumpectomy with pathology revealing a complex cystic mass with fibrocystic changes at 1 to 3 o’clock and intraductal papilloma at 6 o’clock. The patient missed her 6-month follow-up mammogram. She returned 8 months later for a bilateral diagnostic mammogram, which showed a new 2.5-cm mass in the deep central aspect of her left breast at the 12 o’clock position. US showed a cystic mass measuring 3 cm and containing an intracystic solid component measuring 1 × 1 × 2 cm. No axillary or supraclavicular adenopathy was noted on the basis of imaging or physical examination. Her surgical team decided on left breast excisional biopsy with preoperative mammogram guidewire localization. Pathology revealed a benign papilloma measuring 1 cm, focally extending into skeletal muscle in the area adjacent to the previous biopsy site, but with negative margins and no signs of atypia. On the patient’s 6-month follow-up surveillance diagnostic mammogram, another new 3-cm density was noted at the 12 o’clock position. This was most consistent with a benign cyst and was aspirated. She was again lost to follow-up for more than 2 years until July 2015, when she presented with a 2-month history of a slowly enlarging left breast mass in the same region as her previous papillomas. A bilateral diagnostic mammogram with US showed a large mass at the 12 o’clock position measuring 7 × 2.5 cm. Her physical examination revealed that there were two areas of concern: first, a mass measuring 7.5 × 6.3 cm in the 1 o’clock position, and second, an area of nodularity measuring 4.6 × 3.1 cm in the 11 o’clock position. One month later, computed tomography (CT) of the chest and magnetic resonance imaging of the breast revealed a predominantly cystic mass with a solid component extending into the chest wall and approaching the pleural space (Fig. ). These tests also revealed a suspicious internal mammary lymph node (Fig. a). A positron emission tomographic (PET)-CT scan showed a hypermetabolic nodule located in the pretracheal space (Fig. b) with a corresponding standardized uptake value (SUV) of 6.1 and multiple associated hypermetabolic internal mammary lymph nodes with the highest SUV of 6.0 and nodular hypermetabolic activity along the inferomedial aspect of the cystic mass (SUV, 2.7).\nHer case was discussed at the multidisciplinary breast tumor board, and the recommendation was to proceed with a biopsy of the left internal mammary lymph nodes. Core biopsy revealed a papillary neoplasm with benign morphology with immunostains positive for estrogen receptor (ER) at 99%, positive for progesterone receptor (PR) at 85%, HER2/neu 1+, and a Ki67 proliferation index of 6%. An independent external pathologist agreed with the finding of histologically benign papilloma. The patient sustained a biopsy-related internal mammary artery injury and as a result developed a hemothorax requiring video-assisted thoracoscopic surgery.\nUpon recovery from the hemothorax, the patient was referred to the medical oncology department of our hospital. Given the malignant behavior of her tumor, a recommendation of aggressive local control was made. She was started on endocrine therapy with palbociclib and letrozole as a neoadjuvant strategy. Repeat PET-CT following 4 months of combination antiestrogen therapy demonstrated near-complete resolution of metastatic internal mammary lymph nodes (white arrows in Fig. a) and reduced size and avidity of the paratracheal nodes (arrows in Fig. b). A physical examination did not show any significant changes in the size of the left breast mass. She went on to complete 6 months of neoadjuvant therapy. The primary lesion demonstrated minimal clinical response after 6 months of combination endocrine therapy, and then she underwent a left simple mastectomy and sentinel lymph node biopsy. Once again, the pathology revealed a 7.1-cm papillary neoplasm described as microscopically bland and mitotically inactive, with a retained ME layer. Several similar-appearing satellite papillomatous lesions were also seen within the skeletal muscle and deep adipose tissue. Margins and all five sentinel lymph nodes were negative (Figs. and ).\nGiven the extent of her local involvement and history of recurrent disease, she underwent adjuvant chest wall radiation for 6 weeks, followed by adjuvant endocrine therapy with letrozole. Six months after mastectomy, a repeat PET-CT scan showed no evidence of disease. She continues to undergo surveillance CT of the chest and mammography of her right breast. One year after her mastectomy, she underwent left breast reconstruction (Fig. d). She remained without evidence of disease 2.5 years after mastectomy and continued on endocrine therapy during that time. |
A 66-year-old woman was referred to our hospital because of recurrence of pericardial metastatic liposarcoma with progressive dyspnea. The patient had undergone surgical resection of a primary liposarcoma in the left thigh 19 years previously and resection of a locally recurrent tumor 13 years previously. She developed cardiac and pericardial metastasis 10 years later. Resection of a cardiac metastasis originating from the right ventricle was performed via median sternotomy 3 years previously, and a second resection followed by radiotherapy for recurrence of the pericardium metastasis adjacent to the diaphragm 2 years previously. Each of these metastatic tumors was large enough to result in cardiac failure. The pathological diagnosis of the resected primary and metastatic tumors was myxoid liposarcoma. The third recurrence of metastatic pericardial liposarcoma was detected 1 year previously and was followed by metastasis to the posterior mediastinum, liver, retroperitoneum, and peritoneum. Chemotherapy including gemcitabine, docetaxel, pazopanib, and epirubicin was administered for 1 year. Chemotherapy was effective for all of the metastases except those in the pericardium and posterior mediastinum. As the pericardial metastasis enlarged, the patient developed dyspnea over a period of several months. Her symptoms were speculated to be due to diastolic impairment by the large tumor. Reintroduction of chemotherapy was difficult because of cardiac failure. Therefore, she was referred to our hospital for surgical treatment of the pericardial metastasis.\nOn admission, the patient had moderate dyspnea. Signs of cardiac failure, including engorgement of the jugular vein and edema of the lower extremities, were observed. Laboratory data showed an elevated brain natriuretic peptide level of 524 pg/ml. Chest X-ray showed a large mass lateral to the left ventricle extending to the left chest wall. Echocardiography showed a mass lateral to the main pulmonary artery in the pericardium. The left ventricular ejection fraction was preserved (60.9 %), but the inferior vena cava was dilated. Computed tomography showed a large, mostly low-density mass in the pericardium. This mass extended laterally to the ascending aorta and aortic arch, anterolaterally to the main pulmonary artery, and anteriorly to the left pulmonary artery (Fig. ). The mass was speculated to be partially involved in the pulmonary artery. Masses were also identified in the posterior mediastinum, liver, retroperitoneum, and peritoneum. At the time of cardiac catheterization, the right atrial pressure and pulmonary capillary wedge pressure were elevated (11 and 17 mmHg, respectively), and simultaneous measurement of the right and left ventricular pressure demonstrated elevated and nearly equal end-diastolic pressure (16 and 18 mmHg, respectively). The patient’s hemodynamic data corresponded to diastolic impairment. Accordingly, resection of the pericardial tumor was recommended. We performed a median re-sternotomy along the previous skin incision. Moderate adhesion was present between the heart and chest wall, and we safely exposed the right ventricle, right atrium, and ascending aorta (Fig. ). A mucinous mass measuring approximately 30 × 20 × 10 mm, which was not recognized before the operation, was observed between the diaphragm and right ventricle and was resected (Fig. ). The mass and involved pericardium were easily detached from the main and left pulmonary arteries and ascending aorta, and we did not need to use cardiopulmonary bypass. The mass comprised three parts, all of which were mucinous and measured approximately 100 × 50 × 20 mm each. We performed en bloc resection of two of the three masses (Fig. ), but the other was vulnerable and disintegrated. Therefore, we removed them as much as possible. The total weight of the masses was 610 g. The pathological diagnosis of the large masses was myxoid liposarcoma, and that of the smaller mass was myxoid and round-cell liposarcoma (Fig. ). The patient had an uneventful postoperative course, and her symptoms markedly improved. Cardiac catheterization showed a decreased pulmonary capillary wedge pressure of 10 mmHg. Two months later, she underwent resection of the posterior mediastinal metastasis, which had become larger in spite of the chemotherapy, to reduce the tumor volume. This was performed via video-assisted left posterolateral thoracotomy. Chemotherapy was then reintroduced, and the patient was in good condition 3 months after the cardiac surgery. Computed tomography 3 months after the cardiac surgery showed no evidence of recurrence in the pericardium (Fig. ).\nLiposarcoma is the second most common malignancy of soft tissues. The most common primary sites of liposarcoma are the retroperitoneum and lower limbs. Although liposarcoma often metastasizes to different organs, cardiac metastasis, including to the pericardium, is rare. Thirty-five cases of metastatic cardiac liposarcoma have been reported in the literature [–], with only nine cases of pericardial metastases [–]. According to the WHO classification, liposarcoma is divided into the following categories: well-differentiated, differentiated, myxoid, round-cell, pleomorphic, mixed-type liposarcoma, and liposarcoma, not otherwise specified. Myxoid and well-differentiated liposarcomas have more favorable 5-year survival rates than round-cell and pleomorphic types. The first choice of therapy for primary liposarcoma is surgical resection. However, local recurrence or distant metastasis often occurs even many years after surgery. Radiotherapy combined with surgery may be associated with less recurrence []. Chemotherapy is an option in the case of metastatic or unresectable disease [], but it is still an empirical decision owing to the lack of evidence. Pericardial metastasis of liposarcoma often manifests as diastolic cardiac impairment. Surgical resection of cardiac or pericardial metastasis is usually recommended as a potentially radical treatment for solitary lesions without any other metastasis and is occasionally adopted as a palliative procedure in patients with other metastases. In our case, the patient had symptoms of cardiac failure because of impaired diastolic filling caused by the pericardial metastasis. She could not undergo chemotherapy because of cardiac failure. Resection of the tumor was expected to relieve the diastolic impairment and provide a chance of reintroduction of chemotherapy, which could have potentially prolonged her prognosis. Therefore, we decided to perform a third intervention, and the strategy was effective. This is the first reported case of metastatic pericardial liposarcoma, which was successfully resected three times. The prognosis of patients with cardiac metastatic liposarcoma is usually poor. Aoyama et al. [] reported 15 cases of metastatic cardiac liposarcoma; six patients died within 6 months, and the longest follow-up was 15 months. However, the follow-up period of our patient from the first resection of the pericardial metastasis was 51 months. To the best of our knowledge, this is the longest reported follow-up of pericardial metastasis of liposarcoma. Aggressive surgical treatment with the intention of resolving the cardiac impairment may provide a symptom-free interval, and even a prolonged prognosis. |
A 34-year-old male was admitted to the hospital with recurrent episodes of retrosternal chest pain, fatigue, and shortness of breath with an elevated troponin T. He had suffered an acute episode of myocarditis four years previously requiring hospital admission. He had no other relevant medical history and no family history of cardiac disease. He is a nonsmoker and consumed alcohol occasionally. Clinical examination was unremarkable and did not show any evidence of heart failure or systemic disease. ECG showed normal sinus rhythm without any ischemic changes, and chest X-ray showed no evidence of infection or heart failure. Routine blood tests including antinuclear antibody, creatinine kinase (CK), rheumatoid factor, and C-reactive protein were all within normal limits apart from an elevated cardiac troponin T with a peak value of 2700 ng/l (<14 ng/l). Further extensive inflammatory, viral, and autoimmune screening was carried out and found to be negative. Subsequent coronary angiogram showed normal coronary arteries, and transthoracic echocardiography demonstrated left ventricular ejection fraction (LVEF) >55% with trace mitral regurgitation. Cardiac magnetic resonance imaging (MRI) demonstrated extensive subepicardial and midwall late enhancement typical of myocarditis in the anterior, lateral, and inferior walls along with extensive fibrosis with normal LVEF ().\nA short course of steroids and anti-inflammatory medication as an inpatient resulted in the resolution of his myocarditis symptoms. The troponin T level normalized and the patient was discharged with a plan to repeat cardiac MRI in six months. On follow-up as an outpatient, it was decided to refer the patient to rheumatology for an opinion regarding ongoing immunomodulatory therapy. At this juncture, the patient stated that he also had symptoms of stiffness and aching in his calf muscles for quite some time but he did not consider it to be relevant. Despite persistently normal skeletal muscle enzyme levels, an MRI of the lower legs was performed and this showed active myositis involving the gastrocnemius muscles bilaterally (). As the patient was demonstrated to have ongoing myositis despite minimal symptoms, and as he had accrued significant myocardial scarring from previous episodes of myocarditis, it was decided to commence long-term immunomodulatory therapy in the form of methotrexate and prednisolone. Clinically, the patient reported a significant improvement in his symptoms and a repeat of the lower limb MRI demonstrated a significant interval improvement in his skeletal muscle myositis. Six months later, a repeat of the cardiac MRI demonstrated resolution of myocarditis along with persistent, stable, and extensive myocardial fibrosis and preserved LVEF (). The patient is tolerating the immunomodulatory therapy well without major side effects, and he has returned to full-time work. |
A 65-year-old female patient, diagnosed with diabetes mellitus and systemic arterial hypertension, underwent a transesophageal echocardiography (TEE) for a suspected diagnosis of atrial septal defect. The procedure was performed without sedation under topical anesthesia. After three attempts, the procedure was interrupted due to difficulties in progression of the probe into the esophagus, accompanied by retching and an inability by the patient to collaborate with the exam performance.\nAt the end of the examination attempts, the patient immediately experienced neck and oropharyngeal pain. Four hours later she complained of severe dysphagia. On the next day, she noticed edema and hyperemia throughout the anterior neck surface, local pain, and systemic symptoms like weakness and malaise. On the third day she was admitted at the hospital. On physical examination she looked well; her blood pressure was 150/100 mmHg and pulse rate was 100 beats per minute. Edema and hyperemia were evident on the anterior neck surface from the chin up to 3 cm below the furculum where subcutaneous emphysema was also palpable. The oropharynx examination showed intense hyperemia. A computed tomography (CT) scan of the neck showed subcutaneous emphysema in the mediastinal, supraclavicular, and retroesophageal space, which originated from a fistula of the posterior esophageal wall at C6–C7 level ( and ). Upper gastrointestinal endoscopy revealed a marked vascular congestion and edema of the hypopharynx, which progressed to the pyriform sinuses and upper esophageal sphincter, hindering the examination. Passing through the obstruction, just below the cricopharyngeal muscle, a 2 cm perforation of the esophageal wall was evidenced, where a purulent secretion was draining. The remaining endoscopic examination was normal. Facing the diagnosis of esophageal injury with mediastinal involvement, ceftriaxone and clindamycin was initiated and cervicotomy was carried out allowing the drainage of a large amount of pus from the upper mediastinum and periesophageal space. The patient presented with septic shock immediately postoperatively, requiring norepinephrine for hemodynamic stabilization as well as mechanical ventilatory support. After a week of intensive care the clinical status improved with an esophageal fistula remaining. |
A 52 year old right hand dominant male patient, welder by occupation, presented with pain in right shoulder following a fall directly on the right shoulder. Radiographic evaluation revealed Ideberg type III intra-articular glenoid fracture. The patient underwent arthroscopy assisted percutaneous fixation of the fracture.\nThe procedure was performed with the patient in a lateral decubitus position as this facilitated simultaneous arthroscopic as well as fluoroscopic evaluation. The surgeon, the assistants and the scrub nurse were positioned posterior to the patients shoulder. The monitor was positioned on the opposite side at the foot end. The C-arm image was placed in a lateral position across the shoulder with the beam parallel to the floor. The affected limb was positioned in about 40 degree abduction and 20 degree flexion using a traction system to facilitate arthroscopic evaluation. However the traction was kept to minimum to allow manipulation of the arm during fracture reduction\nThrough a standard posterior viewing portal initial diagnostic arthroscopy was performed and the fracture pattern evaluated. The joint was evaluated to rule out other soft tissue injuries. There was fraying of the margin of antero-superior labrum. A 2mm k-wire was passed percutaneously in a superior to inferior direction just adjacent to the Neviaser portal and drilled into the fractured superior glenoid fragment. This k-wire was used as a Joystick to dis-impact the fragment and restore the articular surface under direct arthroscopic vision. An arthroscopic probe introduced through an anterior working portal helped in correcting the rotation of the fracture fragment. Once a satisfactory reduction was achieved, the k-wire was advanced further into the glenoid to obtain provisional fixation.\nA Neviaser portal was then created and a guide wire was passed from superior to inferior direction through the centre of the fracture fragment. The correct placement of the wire was confirmed fluoroscopically. After drilling over the guide wire, definitive fixation and compression of the fracture was achieved with a 35mm partially threaded cannulated cancellous screw. The fracture reduction was again confirmed under both arthroscopic and fluoroscopic visualization. The k-wire used for provisional fixation was then removed and portals closed with simple sutures. The operated limb was put in sling and mobilization was started as tolerated by the patient. The patient was lost to follow up after one month, by which time he was pain free and had a range of movement of 90 degree of forward elevation and abduction. |
A 14 year old boy presented with the complaints of pain, progressive deformity and difficulty in writing using his right hand of 1 year duration. The deformity occurred following a fall from 10 feet height when he sustained an injury of his right forearm. He was initially managed by an osteopath with cleaning, dressing and splint immobilization. No radiographic documentation was carried out at that time. The pain subsided and splint was taken out after 6 weeks. His parents noticed a deformity of the forearm at this time, but did not pursue any further treatment. As the deformity gradually worsened, the child was taken to medical center 4 months after the injury where he was evaluated with radiographs and treated with distal ulnar shortening to correct the forearm deformity []. After 6 weeks of immobilization the child resumed regular activities. He noticed further progression of deformity with pain and difficulty while writing. At this point, he came to our center 1 year after the initial injury. The history was suggestive of punctured wound over the volar aspect on the ulnar side just proximal to the wrist crease, which healed uneventfully with regular dressings. There was no history of persistent infection or wound discharge.\nExamination of the right forearm and hand revealed a manus valgus deformity of 45°. A small puckered scar adherent to underlying structures was noted over the ulnar side just proximal to the wrist crease and no sign suggestive of high grade soft-tissue disruption. A surgical scar was seen over the dorsal aspect of the distal ulna. The ulnar styloid was prominent and extrinsic flexor tightness of 20° (degree) was noted in the fourth and fifth fingers. Wrist dorsiflexion was 5° and palmar flexion was 15° both active and passive. There was a jog of forearm supination and pronation. Shortening of right forearm by 7.5 cm was noted and there was stiffness of the metacarpophalangeal joints and interphalangeal joints. The metacarpophalangeal joints had a jog of passive movement. Active and passive flexion ranging from 20-50° (degree) was noted in the proximal, middle and distal interphalangeal joints.\nErythrocyte sedimentation rate and C-reactive protein were within normal limits. Review of his forearm radiograph carried out before ulnar shortening did not show comminution of radius fracture and there was no ulnar nonunion. The following radiographs revealed a distal radius metaphyseal nonunion with tapered proximal fragment pointing toward the ulna with narrowing of the interosseous space. There was a positive ulnar variance. Due to previous surgery the distal ulna had a nonunion with its epiphysis as well []. Magnetic resonance imaging (MRI) confirmed the diagnosis [] and there were no signs of infection in MRI as well. Preoperative computed tomogram revealed the relative position of the fragments [].\nAfter discussion with the parents and the boy, a preoperative decision was made to do fixator assisted distraction of the forearm bones as a staged procedure to gain length, to be followed later by bone grafting at the site of nonunion. Under general anesthesia monolateral minirail fixator was applied on both radius and ulna percutaneously under image guidance. The nonunion site was not exposed as per the preoperative planning. The parents were taught to do differential distraction and at a rate of 2 mm/day on the radial side and 1 mm/day on the ulnar side from the next day []. Child was followed up with serial radiographs as an outpatient. At the 4th week of distraction the radius had achieved the required length and the ulnar variance got corrected and he was called back for bone grafting. The patient however did not show up at 4 weeks as instructed for bone grafting, but he visited us back at 8 weeks. The repeat radiographs showed new bone formation along the periosteal sleeve of both radius and ulna.\nAt 8 weeks through a volar approach, the bony projection at the proximal end of union site interposing the interossous space was removed as it was hindering the forearm rotation. An intact periosteal sleeve with the new bone at the nonunion site was found and left disturbed. The union site was reinforced with a Kirschner wire []. Biopsy to rule out infection was not done to avoid violating the periosteum and callus at the nonunion site. The fixator was removed along with the Kirschner wire after 3 weeks and a volar splint was used for another 3 weeks and active range of motion therapy was started []. At 1 year followup the boy is pain free and able to do all activities of daily living including writing without pain and the parents were happy with the cosmesis achieved. The child is not willing for any kind of intervention for extrinsic flexor tightness. The preoperative Disabilities of the Arm Shoulder and Hand (DASH) score was 20 and the postoperative DASH score was 28.7 at 1 year followup. Passive palmar flexion had improved to 45° and dorsiflexion to 30° at the end of treatment. Forearm had a passive pronation of 50° and 80° supination []. |
A 45-year-old woman presented to our hospital with multiple lung nodules. She had a history of poorly differentiated thyroid carcinoma, diagnosed 7 months prior to admission, at an outside hospital. The patient was healthy otherwise and reported no radiation exposure or any family history of thyroid cancer. The initial work-up at the time of discovery of the right thyroid nodule included fine needle aspiration and core biopsy, with findings consistent with poorly differentiated thyroid carcinoma. The patient then underwent a total thyroidectomy and central neck lymph node dissection. The pathologic diagnosis from the outside hospital reported a 2.8 × 2.4 × 1.1 cm tumor in the right thyroid without extrathyroidal extension or lymph node metastasis. However, both capsular invasion and extensive vascular space invasion were noted. Based on the tumor size, tumor extension and lymph node status, the tumor was designated as Stage II (pT2 pN0 pMx). IHC staining showed that the tumor cells were positive for thyroglobulin and thyroid transcription factor 1 (TTF1). An immunostain for p53 was also performed at the outside hospital and showed a small focus (< 1 cm) with p53 positivity, suggesting a diagnosis of anaplastic thyroid carcinoma.\nAt our institution, the diagnosis was revised, based on review of both the primary thyroid tumor and the current lung metastases. Both tumors were remarkable for biphasic malignant components: the carcinoma and the sarcoma. The carcinoma component showed a poorly differentiated microfollicular type thyroid carcinoma, composed of sheets and islands of tightly packed thyroid follicles with dense colloid. The tumor nuclei were small and round with vesicular chromatin, resembling those of typical poorly differentiated follicular thyroid carcinoma. Admixed with the epithelial component were malignant spindle cells with small round blue cell type morphology. Focally, rhabdomyosarcoma-like cells with eosinophilic cytoplasm were appreciated. No heterologous cartilage or bone components were identified. The IHC staining performed at the outside hospital showed that the thyroid carcinoma (epithelial) component was positive for thyroglobulin, PAX8 and TTF1 (Fig. ). The sarcoma (spindled) component was negative for all thyroid carcinoma markers (TTF-1, thyroglobulin and PAX8), but was positive for vimentin and focally positive for myogenin (supporting skeletal muscle differentiation) consistent with mesenchymal differentiation. Interestingly, the foci of vascular space invasion contained both epithelial and mesenchymal components as well.\nThe patient received Taxol with Carboplatin for 7 weeks followed by radiation therapy. Her thyroglobulin level rose from 1.2 ng/mL to 25.40 ng/mL 5 months after completion of the chemo-radiation therapy, suggesting progression of the disease. A follow-up CT scan of the chest showed multiple newly developed nodules (ranging from 1 to 2 cm) in the right lung, highly suspicious for metastases. The patient underwent a right thoracotomy, right lung resection/metastasectomy. The surgery was uneventful with negative resection margins. However, the patient’s general condition deteriorated and she succumbed to the disease 4 months later.\nHistological examination of the lung nodules revealed similar tumor morphology and tumor differentiation when compared to the original thyroid tumor, which is somewhat unusual for a biphasic carcinosarcoma (Fig. ). Tumor necrosis was also present. Mutational analysis using a next-generation sequencing based assay showed that the neoplastic cells from the lung metastasis were devoid of genomic alterations for known thyroid cancers, including BRAF, RAS family (KRAS, NRAS and HRAS), EGFR, PTEN, TERT, PI3Kinase or RET. BRAF or RAS family are known as the most commonly altered genes in papillary thyroid cancers. Other molecular mutations reported in the development of anaplastic thyroid carcinoma include p53, PAX8/PPAR gamma rearrangement []. None of the mentioned gene mutations were identified in our patient.\nHowever, an interesting finding in this case is the presence of a point mutation in DICER1 (E1705K) that has previously been associated with differentiated thyroid carcinoma [, ]. Whether the DICER1 (E1705K) mutation is the underlying genetic event leading to the initiation of tumorigenesis or is downstream to other gene alterations in tumor development is largely unknown. Additional mutations of unknown significance were also detected in this tumor including FLCN (R239H), POLD1 (Q684H) and SYK (R217L). These variants have not been adequately characterized in the scientific literature and their prognostic and therapeutic significance is unclear. |
A 66-year-old African-American female with a two-year history of ESRD presumed secondary to longstanding hypertension was in her usual state of health until her HD treatment on the day of admission. She is known to have well-controlled anemia of chronic kidney disease treated with darbepoetin alfa without iron deficiency. Her high blood pressure has been stable on metoprolol tartrate and nifedipine. She had one prior history of congestive heart failure in the setting of fluid overload but no documented history of atherosclerotic cardiovascular disease. She took sevelamer carbonate regularly with her meals to control her hyperphosphatemia. No new medications were added to her medical regimen over the past several weeks prior to her hospitalization. On the day of admission, she had a routine HD treatment start; however within two hours into her regular HD session, she developed sudden onset of diffuse abdominal pain, associated with nausea, and vomiting. Although reportedly the patient remained hemodynamically stable, the dialysis session was immediately interrupted and she was found to have blood infiltration around the arterial needle insertion site of her left upper arm arteriovenous fistula. Her symptoms resolved spontaneously and she went home with her daughter. Within the hour and while at home, the patient started feeling very weak, nauseated and had new onset of red colored urine. Subsequently, she became cyanotic with shortness of breath. Because of these symptoms, emergency medical services were called and she was brought to our emergency department.\nDuring her initial evaluation, she was afebrile and her vital signs were significant for a blood pressure of 154/86 mmHg, a heart rate of 96 beats per minute, respiratory rate of 20 breaths per minute, and oxygen saturation of 97% on 3 liters of oxygen via nasal cannula. Her physical examination was otherwise unremarkable except for bruising and hematoma 2 cm × 2.5 cm over her HD vascular access. Many of the laboratory values obtained in the emergency room were initially limited due to interference (Table ). However, her hematocrit was 10%, with a platelet count of 487,000 per microliter. During her follow-up work-up, the hematocrit level was confirmed to be 10% but the platelet count dropped significantly to 79,000 per microliter and her peripheral smear showed many schistocytes. The patient hemolytic panel was consistent with hemolysis; haptoglobin was less than 30 mg/dL; fibrin split products greater than 20 g/mL and a lactate dehydrogenase of 1,722 IU/L. Prothrombin time and partial thromboplastin time were normal. Her serum potassium was 4 mmol/L. Her fecal occult blood test was negative. Thrombotic thrombocytopenic purpura was considered in the differential diagnosis but ruled out due to the patient’s appropriate response to blood transfusion therapy and lack of neurologic symptoms. Given the patient’s elevated myoglobin, which peaked at 3,101 ng/ml, a rheumatologic workup was ordered and found to be normal. A muscle biopsy showed scattered round, polygonal and angular atrophic myofibers consistent with nonspecific myofiber atrophy. Urine porphobilinogen, serum copper and aluminum levels were all within normal limits. Blood and urine cultures were negative. The patient received packed red blood cells and her hemoglobin and hematocrit rose appropriately. Her hospital course was complicated by an acute myocardial infarction thought to be secondary to supply–demand ischemia in the setting of her profound anemia. She eventually had a full recovery and was discharged from the hospital within one week. Subsequent dialysis sessions were managed with extra care and were uneventful.\nOur patient experienced a severe case of hemolysis associated with HD. This is an uncommon complication of HD but it can be associated with significant morbidity and even death if not recognized early. Hemolysis in this context may be a consequence of a number of biochemical, toxic and mechanical complications during the HD procedure itself. Four major mechanisms capable of inducing hemolysis during HD have been recognized: overheating, hypotonicity of the dialysate, contamination of the dialysate with toxins (e.g., formaldehyde, bleach, chloramine, nitrates) from the water supply, with copper from copper piping and improperly functioning clamps or tubing, such as kinked arterial lines [-]. In cases where there is a contamination of the water supply, most of the patients in the concurrent dialysis session can experience similar signs and symptoms associated with hemolysis []. Toxins can also include contamination of the dialysate with bacteria, endotoxins, and disinfectants although hemolysis due to these toxins is very rare. In one simulated HD study where post HD blood samples were run through faulty tubing, gross hemolysis occurred within thirty minutes [].\nIf faulty tubing was the culprit, one would expect to find sporadic events linked to the manufacturer of these supplies. Further, if biochemical or toxic causes were associated with this event, one would expect to see other patients with similar symptoms and hemolysis. Kinked HD blood lines have been associated with sporadic episodes of hemolysis during HD. However, we believe that this unfortunate event in our patient was induced by malpositioning of the needle on the arteriovenous fistula, particularly given the abrupt onset of symptoms within hours after starting HD, absence of kinked blood lines at the time of the event, presence of blood infiltration with hematoma at the needle insertion site and that this was again a completely isolated event since no other patients suffered this complication. This hemolytic phenomenon is sometimes called “red cell fragmentation syndrome” because the hemolysis is in large part due to intravascular mechanical injury with destruction of the red blood cells.\nOur patient presented with a critical hematocrit level; this seems unique among most HD induced hemolysis cases reported. Other case reports have usually described only minor decrease in hematocrit. This major hemolytic event with hypoxemia may explain the cause of her elevated myoglobin and acute coronary syndrome, which to our knowledge has not been described in other HD-induced hemolysis cases reported. The interference of laboratory values was a result of free hemoglobin that produced a red discoloration of the serum (known as port wine) even after centrifugation. Our patient serum potassium was not elevated, at least in part because the dialysis staff did not reinfuse the patient’s blood back after stopping the HD treatment. |
She is the first child of consanguineous parents and is currently 56 years old. She was born at term after an uncomplicated pregnancy and delivery. The initial developmental milestones were normal. Gait difficulty was noted at the age of 3 years after an episode of febrile seizures. Since then she had progressive walking difficulty which deteriorated over the next 10 years, and was then static during her teenage years. She remained ambulant till the age of 11 years and needed assistance of a walking frame which progressed to wheel chair assistance during the third decade of life. During the course of the illness she received appropriate rehabilitative measures including orthopaedic procedures.\nShe has not had any unprovoked seizures. No learning difficulties were reported and there are no concerns with her vision. Neuropsychological profiling at the age of 47 demonstrated that she functions in the normal range intellectually. She has a psychiatric diagnosis of obsessive compulsive disorder that is currently well controlled on duloxetine.\nOn last follow up at the age of 56 years she was wheelchair dependent. Visual acuity was limited to finger counting close to the eyes. Fundi showed bilateral optic disc pallor. There was lower limb spasticity with bilateral extensor plantar responses. Lower limb lymphoedema was severe, and her extremities were cool and discoloured. She underwent an amputation of the right second and third metatarsals for chronic non-healing ulceration at age 50. Duplex ultrasound and venograms did not show vascular insufficiency. Deep tendon reflexes were brisk except ankle jerks which were bilaterally absent. Sensory system examination showed reduced sensation in both the feet, right foot more affected. There was impaired vibration and position sense. There were no cerebellar signs.\nShe underwent extensive neurometabolic investigations at presentation and the only finding was the persistent elevation in the plasma and CSF glycine levels which were to a lesser degree in comparison to her sister. She was diagnosed as variant non ketotic hyperglycinemia and was treated with protein restricted diet and sodium benzoate to lower her glycine levels which is being continued. Her plasma glycine levels were monitored throughout the course of the illness.\nMRI brain performed at the age of 49 was normal. However, MRI spine showed a focus of high signal in midline posteriorly extending from cranio-cervical junction to level T5-6 (Fig. a, b). |
A 44-year-old woman visited the outpatient clinic with reports of EGD examination showing a GEJ polyp. EGD had been performed as part of a health promotion screening, and the patient denied having any subjective symptom or past medical history including smoking and alcohol consumption. She looked generally healthy. Physical examination and laboratory test revealed that all values were within the normal reference range.\nEGD examination revealed that the polyp was located just on the squamocolumnar junction of the GEJ and presented as a 1.5-cm protruding polypoid mass with an irregular and nodular surface (). Forceps biopsy was performed to exclude malignant tumor. Microscopic examination revealed squamous papilloma showing papillary squamous epithelium with a fibrovascular core. Squamous epithelial cells of the papilloma showed koilocytosis with irregular nuclear membrane and perinuclear halo, which histologically confirmed the presence of human papillomavirus infection ().\nAfter obtaining informed consent from the patient, endoscopic mucosal resection was performed to remove the whole papilloma at the GEJ to avoid a possible increase in the size of the tumor. Histological examination of the resected specimen revealed a nodular mass with papillary growth consisting of squamous cell epithelium with a well-structured inner fibrovascular core, which was compatible with squamous papilloma. The specimen showed complete and curative resection, without involvement of the lateral or vertical margin.\nTwo years later, the patient revisited our clinic for esophageal reflux symptoms. Follow-up EGD incidentally revealed a polyp at the GEJ at the exact spot of the previously removed papilloma. The polyp measured approximately 1.0 cm and had a morphology similar to that of the previous papilloma (), suggesting recurrence. Although the polyp was smaller than that detected 2 years previously, endoscopic submucosal dissection was performed owing to fibrotic scarring and non-lifting sign at the anal side of the polyp (). The polyp was removed en bloc and completely, without marginal involvement. Microscopic examination revealed nodular mass with papillary growth. Histologic examination revealed usual papilloma showing papillary squamous epithelium, with a well-developed fibrovascular core. However, multifocal severe squamous atypia was observed throughout the mucosal epithelium, compatible with squamous cell carcinoma in situ (). The patient was finally diagnosed with squamous cell carcinoma in situ arising from esophageal squamous papilloma. |
A 28-year-old male was referred to our clinic for the management of periodic paralysis (PP). His symptoms first started at age 16. He first noticed significant weakness when he was in the 10th grade. He had gone hunting and had maintained a squatting position for a prolonged period of time (around 45 minutes). When he woke up the next morning, his legs were extremely weak with no associated pain. The weakness was apparently isolated to his legs. During the first workup, his creatine kinase (CK) level was elevated to approximately 600 units per liter (U/L). Over the following month, he slowly regained 75 percent of his prior strength. He denied having back pain and had normal bowel and bladder functions. Shortly after the event, he underwent a neurological assessment. He had two normal lower limb nerve conduction studies (NCS) and electromyographies (EMGs), as well as normal repetitive nerve stimulation. A muscle biopsy taken from the thigh muscle was within normal limits. His CK levels ranged from 600-900 U/L. His weakness remained fairly stable after the initial episode. Four months after the first episode, he went to the beach. He had another episode of weakness during that trip. He did not describe any specific triggers such as hot or cold exposure or specific foods such as carbohydrate ingestion. Between the ages of 14 and 20, he experienced periodic lower limb weakness about once a month. Then he had fewer and fewer episodes until age 28 (2018). Between April of 2018 and October of 2018, he experienced more paralytic episodes, and he did not experience full recovery between those episodes. He had significant weakness for a few days. Then, his strength gradually improved, but he did not return to full strength. He did not have any episodes of dark urine.\nHis past medical history revealed a normal developmental history without any regression. His family history was positive for multiple sclerosis in a paternal uncle and was negative for early muscle weakness and myocardial infarction.\nThe initial physical examination in December of 2018 showed weakness of the bilateral iliopsoas (based on muscle strength grading 4/5) and the adductor magnus (4/5). The remainder of the patient’s neurological examination, including an evaluation of the cranial nerves, a motor exam, a sensory exam and an evaluation of his reflexes, coordination and gait, was within normal limits.\nThe laboratory investigation results, including L-Carnitine levels and a complete metabolic panel, as well as his cell blood count, erythrocyte sedimentation rate, acylcarnitine profile, anti-nuclear antibodies, and his lactate and thyroid levels, were within normal limits. The CK levels ranged from 400-600 U/L.\nHe did not take any medication for PP between the ages of 16 and 28. He initially did not have any cardiac symptoms. The initial electrocardiography (ECG) at age 18 showed multiple ventricular ectopic beats, and the initial echocardiogram was within normal limits. Around age 27, he developed frequent syncope episodes. A cardiac workup showed that his recurrent syncope episodes were related to prolonged QT intervals and polymorphic ventricular tachycardia with deterioration to ventricular fibrillation. A dual chamber, implantable, cardioverter defibrillator was placed which was effective in controlling his syncope episodes. After he developed prolonged QT intervals and ventricular arrhythmia as well as periodic paralysis, a molecular genetic test was performed. The genetic test showed mutations in the KCNJ2 gene. This test was confirmation of a diagnosis of ATS. He was placed on nadolol as well as potassium supplements.\nDuring the neurology visit at age 28, he was put on 250 milligrams (mg) of acetazolamide three times a day. He came back to the neurology clinic after three months for a follow-up visit. He stated that it took a few weeks to see the maximum improvement, but he did not have any more spells, and he regained his full strength. He had always experienced partial improvement between the spells (around 75-80 percent), but this marked the first time that he had experienced full recovery in several years. He tolerated acetazolamide with no significant side effects. The follow-up neurological examination, including cranial nerves, a motor exam, a sensory exam, reflexes, coordination and gait, was unremarkable. The follow-up blood work, including a blood cell count, a comprehensive metabolic panel and CK levels, was within normal limits. |
AT is a 20-year-old Caucasian male who was seen in the emergency department for evaluation of fever, chills, sore throat, and some difficulty swallowing. He was also complaining of mid-back pain, made worse with deep inspiration, and tender swollen glands in his neck. He denied any recent dental procedures or tooth infections. He was seen at his university health center for evaluation five days prior and was diagnosed with viral pharyngitis. He was prescribed a course of prednisone, which he had completed one day prior to presenting to the emergency department. His temperature in the emergency department was 106° Fahrenheit, and his heart rate was 150 beats per minute (bpm). He also complained of some pain in his right calf that he attributed to “slipping and hyperextending his ankle” several days prior. A computed tomography with contrast of his neck was obtained that showed an inflammatory and infectious process of the soft tissue posterior to the oral cavity and extending into the prevertebral soft tissue in the midline. The patient was then transferred to a nearby facility that had otorhinolaryngology coverage for further evaluation. He was admitted to the intensive care unit under sepsis protocol, where he continued to complain of a sore throat in addition to a cough productive of purulent sputum.\nPhysical examination at the time of admission revealed a swollen neck with fullness and tenderness in the anterior and posterior cervical lymph nodes bilaterally. He was unable to open his mouth more than 1.5–2 inches due to pain and swelling. His oropharynx appeared erythematous. He was breathing fast and shallow due to pain with deep inspiration, but he was still able to complete full sentences. He had decreased breath sounds at the bases bilaterally without wheezes or crackles. His right calf was moderately swollen compared to the left and was tender. He had good pulses in both dorsalis pedis and posterior tibialis in both legs in addition to full range of motion in his ankles. Initial lab results showed an elevated white count of 14.1 with 19% bands. His hemoglobin was 15.4, and platelet count was low at 46,000. Creatinine was 1.33, glucose 105, albumin low at 2.6, potassium 3.7, and calcium 8.5. Other lab values of note included alkaline phosphatase 151, aspartate aminotransferase (AST) 46, alanine aminotransferase (ALT) 30, lactate 2.1, and bilirubin 2.6.\nThe treatment plan involved broad-spectrum coverage with administration of intravenous vancomycin and piperacillin/tazobactam, in addition to aggressive IV fluid repletion. The patient was started on low-molecular-weight heparin 40 mg subcutaneous daily for deep vein thrombosis prophylaxis. The chest computed tomography (CT) with contrast showed multifocal areas of necrotizing pneumonia with multiple pulmonary abscesses. A multiloculated effusion was also identified on the left side with a smaller free flowing effusion on the right side. A repeat neck computed tomography (CT) with contrast () showed persistent retropharyngeal and bilateral peritonsillar multiloculated enhancing fluid collections. It also showed thrombosis of the left retromandibular vein and punctate foci within the left internal jugular vein, which was thought to represent partial thrombosis. On the day of admission, the patient underwent computed tomography (CT) guided empyema drainage with placement of left-sided chest tube (). The following day, a second chest tube was placed on the left side and a right-sided chest tube was placed on the third day of hospitalization.\nOn the first day of admission, a right lower extremity ultrasound was ordered due to the pain and swelling in his right calf. This study was negative for deep vein thrombosis. The following day, a magnetic resonance imaging (MRI) of the right lower leg, with and without contrast (), showed edema within the fascial compartments between the gastrocnemius and soleus muscles, as well as in the superficial fascia. A crescent-shaped subfascial fluid collection was also noted over the medial head of the gastrocnemius muscle, indicative of right lower leg fasciitis. The decision was made to proceed with incision and drainage of the fluid collection, in addition to a debridement of the right leg necrotizing fasciitis. Abscess drainage resulted in approximately 500 ml of purulent foul-smelling fluid, some of which was sent for gram stain and culture. A wash out of the right lower extremity wounds would be performed several days later with subsequent placement of negative pressure wound dressing.\nInfectious disease was consulted on the second day of admission. At this time, the patient was being treated with IV cancomycin and piperacillin/tazobactam. The decision was made to add clindamycin to help with possible toxin production and doxycycline due to concern for Brucella and Coxiella. Initial blood cultures had been negative; however, gram-negative anaerobic bacteria were isolated from the fluid sample taken off the right leg abscess. This was later identified to be Fusobacterium necrophorum. On the seventh day of admission, antibiotics were deescalated to meropenem based on susceptibilities. The patient had improved clinically since his admission, but he continued to spike fevers. A follow-up chest CT showed persistence of numerous lung abscesses in the subpleural area with risk of bronchopleural fistula evolution. Due to concern over persistent fevers and empyema, despite multiple chest tube placements, the patient was transferred to a tertiary care center for further evaluation.\nThe patient remained in the ICU at the tertiary care center for another week where he had two more surgeries on his right leg. He also underwent a lung decortication surgery. The patient reported that in his mind, the lung decortication was a turning point in his recovery. He was transferred to a step-down unit and then discharged to home on a four-week course of oral clindamycin. He also underwent outpatient physical therapy. He has since made a complete recovery and was able to continue his undergraduate education. |
A 61 year-old Caucasian woman was referred to our colorectal clinic with an 18-year history of severe intermittent anal pain and constipation. She described experiencing intermittent anal spasms lasting around 15 minutes. These episodes were worse when sitting down for longer than 45 minutes or when lying in bed. The frequency of these anal spasms was increasing with time and occurring every hour at night at the time of presentation. Her constipation symptoms constituted experiencing difficulty in defecation and a sensation of incomplete evacuation. She had no response to amitriptyline or topical diltiazem. Her past medical history was unremarkable apart from four normal vaginal deliveries. Her sister had colorectal cancer diagnosed at the age of 49 and had previously been treated for an undiagnosed anal sphincter problem. There was no other relevant history of note.\nShe initially underwent a flexible sigmoidoscopy and magnetic resonance imaging (MRI) of her perineum. The endoscopy was reported as normal, whereas the MRI showed edema of the IAS. She subsequently had an endoanal ultrasound which confirmed that her IAS was abnormally thick and greater than 5 mm (Fig. ). Anal manometry revealed that although resting and squeeze pressures were within normal limits there were periods of a significant increase in anal resting pressure lasting longer than 2 minutes (Fig. ). Pressures during this period were in excess of 200 mmHg which settled spontaneously. These pressures were even higher than the maximum recorded squeeze pressure (Fig. ).\nShe had an examination of the anal canal under anesthetic which showed a very prominent sphincter complex. She also received Botox injections (Dysport™) at the 3 and 9 o’clock positions of the IAS which led to no subsequent resolution of her symptoms. She then underwent a lateral internal anal sphincterotomy by dividing half of the length (1 cm) of the IAS on the left lateral aspect. A biopsy of the IAS taken at the time of surgery was sent for histology which confirmed polyglucosan body myopathy of the IAS (Fig. ). At 3-month follow-up, she had complete resolution of her symptoms and has not contacted our department with any concerns for more than 1-year postoperatively. |
A 6-year-old male patient was presented in the Department of Oral and Maxillofacial Pathology, National Dental College, Derabassi with a painless left mandibular swelling []. The patient first noticed the swelling one month prior to the presentation. Initially the swelling was minimal but it had grown slowly with time to the present size. The swelling was painless throughout its course.\nExtraoral examination revealed a large irregular swelling in relation to the body of the mandible on left side extending to involve the ramus on the same side. The approximate size of the swelling was 4 cm × 2 cm. The overlying skin was normal in color and smooth.\nOn palpation, the swelling was non tender with bony hard consistency, non compressible, non fluctuant and fixed to the underlying structures. Submandibular lymph nodes were non palpable and non tender.\nIntraoral examination revealed an irregular swelling on the left side of the mandible obliterating the buccal vestibule extending from deciduous first molar to the ramus of the mandible on the same side. The swelling was irregular in shape. The overlying mucosa was normal. Left mandibular first permanent molar had not erupted in the oral cavity.\nRadiographically, orthopantomograph (OPG) showed a radiolucent lesion with scalloped margins on the left side of the mandible involving the body and extending upto the ramus of the mandible, associated with the crown of permanent first molar pushing it against the inferior border of mandible and antero superiorly causing resorption of distal root of 75 [].\nThe lesion was enucleated under general anesthesia and sent for histopathological examination. The tissue received was white in color but in pieces with the largest one measuring 6 cm × 2 cm []. The permanent first molar was also removed along with the tumor.\nThe H and E stained section showed both epithelial and mesenchymal elements []. The mass was composed of embryonic mesenchyme which is traversed by elongated cords of cuboidal and columnar odontogenic epithelial cells that resemble the dental lamina. Some peripheral cells were tall columnar with reverse polarity. Organelle free distal zone was present in these cells. Some follicles containing stellate reticulum in the centre are also identified [].\nMesenchymal component resembled dental papilla. These contain angular cells. There was presence of few delicate collagen fibres.\nThe overall features confirmed the diagnosis of ameloblastic fibroma. |
A 34-year-old male was admitted to the hospital with recurrent episodes of retrosternal chest pain, fatigue, and shortness of breath with an elevated troponin T. He had suffered an acute episode of myocarditis four years previously requiring hospital admission. He had no other relevant medical history and no family history of cardiac disease. He is a nonsmoker and consumed alcohol occasionally. Clinical examination was unremarkable and did not show any evidence of heart failure or systemic disease. ECG showed normal sinus rhythm without any ischemic changes, and chest X-ray showed no evidence of infection or heart failure. Routine blood tests including antinuclear antibody, creatinine kinase (CK), rheumatoid factor, and C-reactive protein were all within normal limits apart from an elevated cardiac troponin T with a peak value of 2700 ng/l (<14 ng/l). Further extensive inflammatory, viral, and autoimmune screening was carried out and found to be negative. Subsequent coronary angiogram showed normal coronary arteries, and transthoracic echocardiography demonstrated left ventricular ejection fraction (LVEF) >55% with trace mitral regurgitation. Cardiac magnetic resonance imaging (MRI) demonstrated extensive subepicardial and midwall late enhancement typical of myocarditis in the anterior, lateral, and inferior walls along with extensive fibrosis with normal LVEF ().\nA short course of steroids and anti-inflammatory medication as an inpatient resulted in the resolution of his myocarditis symptoms. The troponin T level normalized and the patient was discharged with a plan to repeat cardiac MRI in six months. On follow-up as an outpatient, it was decided to refer the patient to rheumatology for an opinion regarding ongoing immunomodulatory therapy. At this juncture, the patient stated that he also had symptoms of stiffness and aching in his calf muscles for quite some time but he did not consider it to be relevant. Despite persistently normal skeletal muscle enzyme levels, an MRI of the lower legs was performed and this showed active myositis involving the gastrocnemius muscles bilaterally (). As the patient was demonstrated to have ongoing myositis despite minimal symptoms, and as he had accrued significant myocardial scarring from previous episodes of myocarditis, it was decided to commence long-term immunomodulatory therapy in the form of methotrexate and prednisolone. Clinically, the patient reported a significant improvement in his symptoms and a repeat of the lower limb MRI demonstrated a significant interval improvement in his skeletal muscle myositis. Six months later, a repeat of the cardiac MRI demonstrated resolution of myocarditis along with persistent, stable, and extensive myocardial fibrosis and preserved LVEF (). The patient is tolerating the immunomodulatory therapy well without major side effects, and he has returned to full-time work. |
A 22-year-old Sri Lankan woman was referred from the orthopedics unit to the endocrinology unit, National Hospital of Sri Lanka, for further evaluation of recurrent femur fractures. She first experienced a right sided subtrochanteric fracture following a low trauma injury at the age of 20 years (), for which she underwent internal fixation. Thereafter, she experienced a left intertrochanteric femur fracture at the age of 22 years, 2 months prior to the current presentation.\nShe had an interesting past medical history. She was diagnosed to have a left side nephroblastoma at the age of 8 years, for which she underwent left side radical nephrectomy followed by chemotherapy. Thereafter, at the age of 14 years, she developed a right sided facial swelling and was found to have a maxillary tumor which was described during the surgery as a well-circumscribed mass lesion in the left maxilla, eroding the anterior wall of maxilla and lateral nasal wall. She underwent partial maxillectomy for this, and histology revealed features suggestive of fibrous dysplasia.\nShe did not have a history of chronic steroid use, hypogonadism, hyperthyroidism, rheumatoid arthritis, and exposure to cigarette smoking to suggest secondary causes for osteoporosis. She had neither any features of malabsorption nor inadequate exposure to sunlight to suggest a vitamin D deficiency causing these fractures. The histology of bone biopsies was negative for metastatic deposits.\nShe did not have a family history of hyperparathyroidism, renal, or uterine tumors. There was no past history or family history suggestive of MEN 1. Her general and systemic examinations were normal apart from the healing fracture sites.\nHer investigation findings are summarized in .\nHer investigations revealed high total calcium and urinary calcium excretion, with low serum phosphate level. She was found to have a vitamin D deficiency, which was corrected by vitamin D replacement. As high calcium and low phosphate were suggestive of a primary hyperparathyroidism, intact parathyroid hormone (PTH) was done after correcting vitamin D level, which was found to be very high at 1025 pg/ml (10–65 pg/ml), confirming the diagnosis of hyperparathyroidism.\nHer left forearm dexa scan revealed osteoporosis with a Z score of −5.6, suggestive of active chronic hyperparathyroidism, leading to bone resorption and reduced bone mineral density with preferential involvement of cortical bone and increased risk of fracture [].\nThree-dimensional computed tomography (CT) of neck was carried out for localization, which revealed a 2.5 × 2.9 cm well-defined rounded mass with avid contrast enhancement posteroinferior to the left lobe of the thyroid gland, suggestive of a parathyroid mass (). Nuclear imaging was not done as the facility was not available at our setting.\nAt this point, due to the constellation of nephroblastoma, maxillary tumor, and primary hyperparathyroidism with a parathyroid mass, a possibility of hyperparathyroidism jaw tumor syndrome was suspected although a suggestive family history was not there. Therefore, the genetic studies were carried out via target region capture followed by next generation sequencing, using a specific indel detection tool, which revealed a positive CDC73 mutation with a large deletion of exon 1–17. This mutation was verified by reverse transcription polymerase chain reaction (RT-PCR), where exons 1, 10, and 17 were selected for verification. This confirmed the diagnosis of hyperparathyroidism jaw tumor syndrome (). MEN 1 and calcium sensitive receptor mutations were negative.\nThe patient underwent subtotal thyroidectomy with parathyroidectomy with removal of left lower parathyroid mass, which was a smooth, round, well-demarcated mass (), and the remaining parathyroid glands were atrophied.\nImmediate postoperative PTH level was reduced by more than 50%–76 pg/ml, suggesting successful removal of the parathyroid mass. The histology revealed encapsulated parathyroid adenoma composed of a proliferation of polygonal cells separated by fibrovascular septa, with round to ovoid nuclei with dispersed chromatin and inconspicuous nucleoli and moderate eosinophilic cytoplasm. Mitoses were rare, and there were also cystic areas. There was no evidence of vascular invasion, necrosis, or invasion into soft tissues of thyroid gland. These features were compatible with a parathyroid adenoma.\nCurrently, the patient is on calcium and calcitriol treatment and being followed up with serum calcium and phosphate levels with monitoring of urinary calcium excretion. Her imaging of the abdomen did not reveal a recurrence of nephroblastoma or uterine tumors. Biochemical screening of family members was negative, and genetic screening is underway. |
A 61 year-old Caucasian woman was referred to our colorectal clinic with an 18-year history of severe intermittent anal pain and constipation. She described experiencing intermittent anal spasms lasting around 15 minutes. These episodes were worse when sitting down for longer than 45 minutes or when lying in bed. The frequency of these anal spasms was increasing with time and occurring every hour at night at the time of presentation. Her constipation symptoms constituted experiencing difficulty in defecation and a sensation of incomplete evacuation. She had no response to amitriptyline or topical diltiazem. Her past medical history was unremarkable apart from four normal vaginal deliveries. Her sister had colorectal cancer diagnosed at the age of 49 and had previously been treated for an undiagnosed anal sphincter problem. There was no other relevant history of note.\nShe initially underwent a flexible sigmoidoscopy and magnetic resonance imaging (MRI) of her perineum. The endoscopy was reported as normal, whereas the MRI showed edema of the IAS. She subsequently had an endoanal ultrasound which confirmed that her IAS was abnormally thick and greater than 5 mm (Fig. ). Anal manometry revealed that although resting and squeeze pressures were within normal limits there were periods of a significant increase in anal resting pressure lasting longer than 2 minutes (Fig. ). Pressures during this period were in excess of 200 mmHg which settled spontaneously. These pressures were even higher than the maximum recorded squeeze pressure (Fig. ).\nShe had an examination of the anal canal under anesthetic which showed a very prominent sphincter complex. She also received Botox injections (Dysport™) at the 3 and 9 o’clock positions of the IAS which led to no subsequent resolution of her symptoms. She then underwent a lateral internal anal sphincterotomy by dividing half of the length (1 cm) of the IAS on the left lateral aspect. A biopsy of the IAS taken at the time of surgery was sent for histology which confirmed polyglucosan body myopathy of the IAS (Fig. ). At 3-month follow-up, she had complete resolution of her symptoms and has not contacted our department with any concerns for more than 1-year postoperatively. |
A 23-year-old French man of African origin, an elite football player, sustained a midshaft anterior cortex tibial stress fracture 2.5 years ago. Initially, he was treated with cast immobilization, no weight bearing for 3 months, ultrasound stimulation, and electromagnetic field therapy. The fracture did not heal; he had pain during gait, so he continued no weight bearing for 3 additional months. After that period, the fracture site still was not healed, so he underwent an operation performed by his team doctor. In this operation, the medullary canal of his tibia was reamed and an im nail was inserted.\nUnfortunately, the fracture site did not consolidate again, even 18-months postoperatively, so he presented to our clinic for counseling. It was obvious from the X-ray (Fig. ) that a nonunion of the fracture had occurred.\nHe did not smoke tobacco and he had a free medical history. When he presented to our clinic, the area at the fracture site was swollen and painful when palpated. The pain got worse when he attempted to walk with full weight bearing, so he had to use crutches. An examination of the peripheral nervous system of his lower extremities did not provide us with any pathologic findings. In addition, the laboratory examinations for possible endocrine or metabolic disorders were negative (Table ), so he was advised to have a reoperation to address this nonunion. The treatment options for such cases include nail exchange, drilling of the fracture site, bone grafting, or removal of the nail and internal fixation with a plate. We performed a tension band plate fixation, which is a technique already described for the treatment of anterior tibial stress fractures that failed non-operative treatment [], with bone grafting and without removing the nail.\nA longitudinal incision was made just lateral to the anterior tibial crest centered over the fracture site. The fascia over the tibialis anterior was divided, the muscle lifted off and the fracture site was visualized. The necrotic bone and callus at the fracture site was debrided with the use of an osteotome and a curette. Transverse drilling around the fracture site was done to promote healing and osteoblastic activity. Bone marrow from the ipsilateral iliac crest was inserted into the fracture site and a tension band plate was applied over the im nail.\nWe used a 6-hole, 4.5 mm locking compression plate. The plate was prebended and the screws were placed in a compression manner to achieve a tension band effect to the fracture site. A cortical screw was put first to the distal hole closest to the fracture site and then a cortical screw to the closest hole proximal to the fracture site to ensure compression of the fracture. Consequently, one unicortical locking screw was inserted proximally to the fracture site and the other two distally. With the use of locking and non-locking screws we minimized the pressure at the periosteum, which can damage blood supply to the poorly vascularized bone. The screws were angled in a different axis in order to bypass the nail (Fig. ).\nPostoperatively, our patient was advised to wear an orthotic boot and to not bear weight for 6 weeks. Range of motion exercise involving knee and ankle and isometric exercises were initiated immediately postoperatively. After 6 weeks he progressed to weight bearing as tolerated. At 3 months postoperatively he was pain free and started light jogging, swimming, and plyometric and core stabilization exercises. At 6 months postoperatively the complete radiologic union of the fracture was evident (Figs. and ). He was symptom free; he resumed at that time a full training program and he returned to play football 6 months postoperatively at his preinjury high competition level. |
The patient is a 39-year-old gentleman originally from Central America who currently lives in Southeast Georgia. His past medical history is significant for HIV/acquired immunodeficiency syndrome (AIDS), alcohol abuse, tobacco dependency and bone marrow biopsy-proven leishmaniasis who was transferred from an outside hospital. He denied any recent travel. At first presentation, the patient had diffuse moderate abdominal pain, nausea, vomiting, chills and dysphagia. On physical exam, the patient was tachycardic to a rate of 140 beats per minute and the patient was found to have mild abdominal tenderness, but the physical exam was otherwise non-contributory. The patient was leukopenic as well as thrombocytopenic. For treatment, the patient was placed on amphotericin B for his leishmaniasis as well as highly active antiretroviral therapy (HAART) for his HIV/AIDS infection. The patient was then discharged home on a 3 month course of amphotericin B in addition to continued HAART therapy with outpatient follow-up.\nOver the course of the next 6 months, the patient initially recovered from his acute illness, but later developed acute abdominal pain and tenderness along with nausea, which prompted presentation to the emergency department. On exam, the patient was found to have a fine nodular skin rash across his chest, abdomen and parts of his back consistent with cutaneous leishmaniasis. Additionally, he was found to have continued thrombocytopenia. A bone marrow biopsy was done to differentiate whether the new onset symptoms were secondary to his HIV or related to a relapse of the his leishmaniasis. The bone marrow results are shown in \n, which demonstrates various stains showing the presence of macrophages filled with Leishmaniasis amastigotes. The patient was again initiated on intravenous amphotericin B for 6 weeks inpatient and discharged with the plan to get an amphotericin B infusion every 3 weeks on an outpatient basis. The patient was steadily improving on his inpatient treatment and was cleared for discharge home, the patient, however, was lost to follow-up.\nSeveral years later the patient returned to the emergency department with the most recent flare of his Leishmaniasis disease. The patient presented with rectal bleeding accompanied by continued abdominal pain and discomfort. This hospital course, the gastroenterology team was consulted. An esophagogastroduodenoscopy was performed, and a biopsy was taken of his gastric mucosa. The biopsy showed scattered macrophages filled with amastigotes throughout the patient’s gastric mucosa (see\n). These findings confirmed the suspected case of gastric leishmaniasis. The patient was subsequently treated with amphotericin B to treat his disseminated gastric leishmaniasis and encouraged to stay consistent with his HAART for his HIV. The patient responded well, his gastrointestinal bleed resolved, and the patient clinically had no complaints or concerns. The patient was encouraged to follow-up in the outpatient setting with his gastroenterologist for his gastric leishmaniasis and primary care physician for his HIV/AIDS disease. |
A 46-year-old female with a known history of hypothyroidism for 10 years presented in the outpatient department with dry cough for one week, fever for two days and breathlessness for 10 days which has worsened over the last two days. She had no history of power loss in any of her limbs. She had no history of smoking. She had no family history of any autoimmune disorders.\nShe visited a primary care physician one day earlier and was recommended arterial blood gas evaluation to detect the degree of respiratory impairment and possible etiologies. The arterial blood gas analysis report showed a pH of 7.3, pCO2 of 60 mmHg, pO2 of 52 mmHg and the patient was advised admission considering a high level of pCO2. On admission, blood samples were drawn for complete blood count, renal function test, liver function test, and thyroid stimulating hormone; all of which showed normal results. To rule out the pulmonary thromboembolism, a cardiothoracic pulmonary angiogram was performed which showed posterobasal consolidation in both lower lobes with a thin rim of pleural effusion bilaterally and a few enlarged homogeneously enhancing lymph nodes in the perivascular and right paratracheal regions.\nOn admission, the patient was also started on non-invasive positive pressure ventilation, bronchodilators, and broad-spectrum antibiotics against pneumonia. Despite these interventions, the patient showed no improvement for the next 24 hours. The subsequent arterial blood gas analysis showed a pH of 7.2 with pCO2 of 105 mmHg. Breath holding counts decreased from 12 to 6, thus developing hypercapnia. In addition, the patient started developing drowsiness for which a neurology consultation was ordered to rule out other neurologic causes of type 2 respiratory failure. To rule out myasthenia gravis, various serological tests were performed including acetylcholine receptor antibodies, IgA antibodies, electromyography with the results expected to arrive the next day.\nOwing to the expected delay in the results, a bedside ice pack test was performed to diagnose myasthenia gravis. A positive response was elicited with the test (Figure ) and neostigmine was started bearing in mind the deteriorating condition of the patient. Within 3 hours, pH was 7.3, pCO2 was 75 mmHg, pO2 was 70 mmHg with a gradual lowering of the pCO2. Her consciousness gradually improved and the non-invasive ventilator support was reduced within 6 hours. Next day all the results of the serological tests were obtained and turned out to be negative for myasthenia gravis. However, the electromyography test result was positive for two muscles confirming the diagnosis of myasthenia gravis. Detailed laboratory findings and investigation reports are shown in Table . |
A 23-year-old French man of African origin, an elite football player, sustained a midshaft anterior cortex tibial stress fracture 2.5 years ago. Initially, he was treated with cast immobilization, no weight bearing for 3 months, ultrasound stimulation, and electromagnetic field therapy. The fracture did not heal; he had pain during gait, so he continued no weight bearing for 3 additional months. After that period, the fracture site still was not healed, so he underwent an operation performed by his team doctor. In this operation, the medullary canal of his tibia was reamed and an im nail was inserted.\nUnfortunately, the fracture site did not consolidate again, even 18-months postoperatively, so he presented to our clinic for counseling. It was obvious from the X-ray (Fig. ) that a nonunion of the fracture had occurred.\nHe did not smoke tobacco and he had a free medical history. When he presented to our clinic, the area at the fracture site was swollen and painful when palpated. The pain got worse when he attempted to walk with full weight bearing, so he had to use crutches. An examination of the peripheral nervous system of his lower extremities did not provide us with any pathologic findings. In addition, the laboratory examinations for possible endocrine or metabolic disorders were negative (Table ), so he was advised to have a reoperation to address this nonunion. The treatment options for such cases include nail exchange, drilling of the fracture site, bone grafting, or removal of the nail and internal fixation with a plate. We performed a tension band plate fixation, which is a technique already described for the treatment of anterior tibial stress fractures that failed non-operative treatment [], with bone grafting and without removing the nail.\nA longitudinal incision was made just lateral to the anterior tibial crest centered over the fracture site. The fascia over the tibialis anterior was divided, the muscle lifted off and the fracture site was visualized. The necrotic bone and callus at the fracture site was debrided with the use of an osteotome and a curette. Transverse drilling around the fracture site was done to promote healing and osteoblastic activity. Bone marrow from the ipsilateral iliac crest was inserted into the fracture site and a tension band plate was applied over the im nail.\nWe used a 6-hole, 4.5 mm locking compression plate. The plate was prebended and the screws were placed in a compression manner to achieve a tension band effect to the fracture site. A cortical screw was put first to the distal hole closest to the fracture site and then a cortical screw to the closest hole proximal to the fracture site to ensure compression of the fracture. Consequently, one unicortical locking screw was inserted proximally to the fracture site and the other two distally. With the use of locking and non-locking screws we minimized the pressure at the periosteum, which can damage blood supply to the poorly vascularized bone. The screws were angled in a different axis in order to bypass the nail (Fig. ).\nPostoperatively, our patient was advised to wear an orthotic boot and to not bear weight for 6 weeks. Range of motion exercise involving knee and ankle and isometric exercises were initiated immediately postoperatively. After 6 weeks he progressed to weight bearing as tolerated. At 3 months postoperatively he was pain free and started light jogging, swimming, and plyometric and core stabilization exercises. At 6 months postoperatively the complete radiologic union of the fracture was evident (Figs. and ). He was symptom free; he resumed at that time a full training program and he returned to play football 6 months postoperatively at his preinjury high competition level. |
A healthy 45-year-old Caucasian female presented with swelling and discomfort in the upper right posterior area, which had persisted since the extraction of her upper right second premolar. The tooth had been restored with a large amalgam []. When it fractured ten months earlier, it was deemed non-restorable. The tooth had a mesial root curvature and required surgical extraction. Immediately after the extraction, the adjacent teeth were prepared for a fixed prosthesis and a temporary bridge was fabricated. No postoperative radiographs were exposed during this appointment.\nThe patient returned the following day with pain, swelling, and facial bruising. Her dentist prescribed ibuprofen and cold compresses, but the swelling failed to resolve. When symptoms persisted to the eleventh post-operative day, her dentist removed the temporary bridge, curetted the extraction site, and prescribed clindamycin. When she was re-evaluated on the twenty-third postoperative day, there was continued swelling, but wound healing appeared to be progressing. Within three months after the extraction, the site was covered by intact keratinized tissue. Although the swelling had not resolved, the permanent bridge was completed and cemented.\nThe patient presented to our practice seven months later for investigation of the swelling. Radiographs revealed a radiopaque object distal to the upper right first premolar and a mesial osteotomy defect []. A segmental osteotomy performed thirty years earlier to correct a maxillary midline discrepancy had failed to heal completely. The edentulous site exhibited firm facial swelling, but did not appear granulomatous. After obtaining local anesthesia, a facial full thickness flap was reflected from the distal of the first molar to the distal of the canine. An encapsulated acrylic object and associated granulation tissue were removed []. Severe bone loss was noted distal to the first premolar. Since the site was inflamed and access was limited by the bridge, no bone graft was placed prior to wound closure. At a follow-up appointment one month later, there were no signs of abnormal healing. However, the gingival margin on the distofacial and distal aspects of the premolar had receded 2.5 mm in conjunction with tissue shrinkage at the edentulous site.\nFinal healing was evaluated eight months later. Probing depths were acceptable and inflammation was largely absent. Gingival recession previously noted around the first premolar had not progressed. A radiograph suggested that the density of the bone distal to the first premolar had increased []. Given these findings, it was feasible for the patient to retain the bridge. |
A 63-year old female presented with weakness of her left leg. She had been treated in 2001 for a superficially spreading melanoma, Breslow depth 1.4 mm. Magnetic Resonance Imaging (MRI) of the brain revealed a metastasis in the right frontal lobe with signs of hemorrhage and several additional small cerebral metastases. Subsequent computed tomography (CT) scans showed metastases to the thoracic and lumbar spine. A biopsy of a metastasis at the sacro-iliac joint revealed melanoma cells; mutation analysis of the BRAF gene showed a V600E mutation in exon 15. Initial treatment consisted of whole-brain radiation (7×4 Gy), and radiation to the thoracic and lumbar spine. Since all of the known metastases had been treated with radiation, systemic treatment was not initiated yet.\nA CT scan made two months later revealed new metastases in the right lung, peritoneum and left groin. The patient had recovered well from the cerebral hemorrhage and the treatment of her cerebral and spinal metastases. She was able to walk for a short distance and her only complaint was a moderate hearing loss. MR imaging of the brain revealed a slight decrease of the cerebral hemorrhage and no new metastases (Figure A). Vemurafenib, an oral inhibitor of the BRAF kinase, was initiated at 960 mg bi-daily. Treatment was initially tolerated well except for mild periorbital edema.\nAfter seven weeks of treatment with vemurafenib, she presented to the hospital with severe visual loss, which had started several days earlier. She did not have a previous medical history of ocular problems. An MRI of the brain showed less hemorrhage of the right frontal metastasis and no increase in size of the other small cerebral lesions (Figure B). A CT scan showed regression of the peritoneal and pulmonary lesions and stabilization of the metastasis to the right groin. Ophthalmological examination revealed a visual acuity of only light perception in both eyes. Slit lamp examination showed shallow anterior chambers in both eyes, and a severe fibrinous and cellular reaction, covering the entire pupillary opening and causing a pupillary block and secondary elevation of the ocular pressure (Figure ). Ultrasound imaging of the eyeball showed signs of scleritis. Vemurafinib was considered the culprit and therefore discontinued; treatment with topical and systemic coricosteroids (prednisone, 60 mg per day) was initiated. The patient’s scleritis decreased and her vision improved slowly to a visual acuity of 0.25 in the right and 0.8 in the left eye. At that time, fundoscopic examination was possible, and did not reveal signs of vasculitis nor chororetinitis in both eyes. A surgical peripheral iridectomy was performed in the right eye to reverse a pupillary block caused by posterior synechiae.\nFour weeks after cessation of treatment, she presented with progressive aphasia. An MRI of the brain showed progression of cerebral metastases with new hemorrhages in several metastases (Figure C). At that moment, her vision had improved, but had still not fully recovered. Because of the severe impact of the visual loss on quality of life, and since the response of the cerebral metastases at 7 weeks of treatment with vemurafenib showed stabilization at best, a second attempt of treatment with BRAF inhibitors was not initiated. Second line treatment with ipilimumab, an anti-CTLA4 antibody, was considered. The occurrence of a severe pan-uveitis was judged to be a contraindication to therapy that acts by stimulating the immune system. Additionally, she was still being treated with systemic corticosteroids. Dacarbazine was considered, but viewed as a treatment with little chance of response in this setting. The patient and her family preferred to refrain from further systemic treatment of her cancer. She died at her home six weeks later. |
An 80-year-old male patient was admitted to the emergency medical center of our hospital based on a complaint of myalgia and abdominal pain. According to the patient and his daughter, the patient had no known comorbidities, including psychiatric disorders, immune deficiency or trauma-related problems. The patient was hemodynamically unstable; he was hypotensive, and his body temperature was increased to 40.7 °C upon the first examination. As the patient exhibited jaundice with abnormal laboratory findings (total bilirubin, 5.46 mg/dL; aspartate transaminase, 251 U/L; alanine transaminase, 143 U/L), bedside biliary ultrasonography was immediately performed, and the results showed no abnormal findings with regard to the biliary system. A subsequent computed tomography (CT) scan of the abdomen showed diffuse wall thickening and several diverticula of the sigmoid colon with multiple air bubbles in the portal venous system (Fig. a and b). Nevertheless, we found no abnormalities in the biliary system, such as a gallstone or cholelithiasis; however, no further information could be established via the CT scan because the study was performed without contrast enhancement due to the decreased renal function of the patient. The initial diagnosis made at the time of admission was septic shock caused by atypical biliary disease; therefore, this patient was admitted to our surgical intensive care unit and received fluid resuscitation and empirical antibiotic treatment (meropenem against suspicious gram-negative bacteremia), although we could not determine the definitive infectious source of septic shock in this patient.\nAfter admission to the intensive care unit and 5 days of hospitalization, the patient became hemodynamically stable, and the laboratory findings of jaundice and renal function were improved; however, he complained of persistent abdominal pain. As the renal function of the patient improved, we performed a consecutive abdominal CT scan with contrast enhancement to identify any possible cause of the abdominal pain in this patient and to recheck for any infectious origin of the sepsis. In contrast with the initial CT scan, the second CT scan showed a 5 cm abscess in the mesentery of the sigmoid colon and aggravated pylephlebitis of the portal venous system (Fig. a and b). Additionally, Escherichia coli (E. coli) bacteremia was confirmed via a blood culture test performed at the time of admission; therefore, the diagnosis of this patient was changed to sigmoid colon diverticular perforation with pylephlebitis based on both initial and consecutive CT scans. We performed sigmoidoscopy to identify the diverticula of the sigmoid colon; however, no diverticular disease was found (Fig. ). As the results of the diagnostic work-up were conflicting and fatal outcomes were expected due to persistent pylephlebitis and abdominal pain, we performed an exploratory laparotomy on the 8th hospital day. The abdominal cavity was entered via a low midline incision. The peritoneal cavity was clean with no contamination, and we could not find any perforation or abnormalities in the biliary system or intestine, including the sigmoid colon. Instead, we observed an extraordinary retroperitoneal bulge, to which the sigmoid colon mesentery was attached, and retroperitoneal dissection revealed a 5 × 5 cm whitish abscess pocket around the iliac artery bifurcation (Fig. ). This abscess cavity was completely separated from the sigmoid colon mesentery, and the capsule was severely adhered to the aorta and left iliac artery. The capsule was a solitary abscess with no fistula with adjacent organs. As the complete removal of the abscess cavity required massive dissection and might have caused unpredictable complications, we performed only incision, drainage, and curettage. A microbial culture test was also performed on the tissue and pus of the abscess cavity. Finally, an additional examination of the whole peritoneal cavity and organ systems was conducted; however, no infectious origin of the RA was found in the gastrointestinal or genitourinary system.\nAfter the operation, microbial blood culture tests were performed, and intravenous vancomycin was empirically added to the antibacterial regimen to cover methicillin-resistant Staphylococcus aureus (MRSA), which is one of the major pathogens isolated from RAs. After a detailed interview with his daughter, we found that this patient had received frequent acupuncture by an unqualified therapist in the low lumbar region to treat chronic back pain, and the point of the acupuncture was exactly identical to that of the RA. The clinical and laboratory parameters of the patient gradually improved, and oral feeding was resumed on the 4th postoperative day. MRSA was identified in the microbial culture tests of pus and tissue; however, a blood culture test performed immediately after the surgery revealed no pathogen. In addition, there was no evidence of tuberculosis or malignancy on pathological examination for retrieved tissue. The patient responded well to the treatment and was discharged from the hospital at 20 days after the operation. We checked a follow-up CT scan taken in the outpatient department 40 days after the operation and identified no remaining pylephlebitis or abscess. |
We present a case of 40-year-old building and construction male worker who slipped and fell from a height of three (3) meters and sustained a deep penetrating wound on the right side of the anterior neck a week prior to presenting at our facility. He was apparently working from the above height when he slipped and fell on a sharp piece of iron rod which penetrated deep into the right anterior neck. He quickly pulled the sharp iron rod out when he got up from the floor. According to him, the bleeding was not profuse and stopped when he arrived at the local hospital to search for remedy (). He did not have hemiplegia, paraplegia, or quadriplegia when we saw him. He is not known to be hypertensive. He did not take alcohol prior to the fall although he takes alcohol occasionally. He had a left femoral fracture at the age of 24 and a right femoral fracture at the age of 32; both incidences were operated on successfully. On examination at our facility we saw a middle aged man who was conscious and alert but however acutely ill with his neck fixed in cervical collar. General as well as systemic examination did not yield much. All the systems where grossly normal. Neurological examination revealed normal pupils which reacted normally to light. Cranial nerves examination was unremarkable. Power on four limbs as well as reflexes was normal. Digital rectal examination revealed a normal spinster tone. Routine laboratory as well as other ancillary (ECG, CXR, etc.) investigations were normal.\nNeck CT-scan done at the local hospital revealed C2-C4 transverse process fractures on the right side, fracture at the right lamina of C3, and right common carotid artery dissection. CT-scan of the head showed no abnormalities (Figures and ). Explorative three-dimensional reconstruction plain and enhanced scan imaging of the cervical spine, chest, and abdomen done at our facility revealed two segmental stenoses of the right common carotid artery with very pale V1 and V3 segment of the right vertebral artery as well as blockage at V2 segment (Figures –) as well as fracture at the right lamina of C3 and C2-C4 transverse processes with free bone fragments and peripheral soft tissue swelling (Figures –). The skin at the right anterior cervical region is discontinuous, with adjacent soft tissue swellings and gas accumulation. The bilateral carotid artery sheath lymph nodes slightly enlarged. At the upper lobe of the right lung there were multiple calcifications, some of which were adjacent to the pleura. There was also slight thickening of the left pleura. The heart was not enlarged but we observed slight accumulation of gas in the anterior mediastinum. Multiple low-density lesions were seen in the liver which we think are constant cysts. A working diagnosis of right common carotid artery dissection with C1-C4 fractures was made.\nAfter preoperative education and counselling of the patient as well as the relatives, surgery was scheduled the next day. Intraoperative cerebral angiography showed right carotid artery dissection and right vertebral artery occlusion. There was some reparation at the distal end of the right vertebral artery. The left vertebral artery was however normal. We introduced the guiding catheter guide wire to the proximal end of the right common carotid artery with continued infusion of heparinized saline, after which we introduced a guide wire with a Cordis stent (10 ∗ 60mm) to completely cover the right common carotid artery dissection site with stenosis and released the stent gradually until it completely filled the stenosis area (Figures –)). We delivered contrast agent into right common carotid artery to make sure it was patent before removing the guiding catheter followed by withdrawal of the femoral arterial sheath. Control contrasted angiograph done revealed stenting was successful (Figures and ). The patient recovered markedly and was discharged home a week after. Scheduled outpatient visit every 6 months for 2 years revealed no neurological complications. |
This case report describes a 33-year-old female currently undergoing breast cancer treatment following the AC-T-T (doxorubicin hydrochloride (Adriamycin) and cyclophosphamide followed by paclitaxel (Taxol) and trastuzumab (Herceptin)) treatment regimen at the University of Texas MD Anderson Cancer Center in Houston, Texas, USA. She reported an episode with profuse spontaneous bleeding located at the palatal gingiva in the maxilla between the left central and lateral incisor. The patient had no other known medical or oral conditions except for breast cancer, which was treated with 12 weekly infusions of paclitaxel and trastuzumab (T-T) every three weeks prior to her scheduled mastectomy. Her last infusion of T-T was six days prior to the incidence of the intraoral bleeding. The patient reported spontaneous bleeding from her mouth as she was getting dressed in the morning, which occurred before brushing her teeth or receiving any oral stimulus that may cause gingival bleeding, and became very concerned as the bleeding was profuse and difficult to control. A complete clinical oral examination was performed by a periodontist within an hour of the bleeding incident. The probing depths of teeth adjacent to the bleeding site were within normal limits, ranging from 2 to 3 mm, and no visible plaque was detected. A periapical intraoral radiograph from the anterior maxillary area revealed no signs of bone loss or other signs of pathology (, , and ). The patient has no previous history of periodontal disease, no trauma to the area and no para-functional habits, excellent oral hygiene, and there was no evidence of increased pocket probing depths or gingival inflammation or infection noted. There were neither reports of pain nor discomfort. The patient reported previous episodes of minor epistaxis that started after initiating treatment with the PT regimen, but no prior history of gingival bleeding. The patient was also not menstruating at this time. The patient started to bleed profusely again upon examination and gentle probing of the gingiva by the periodontist. The bleeding source was located at an area in the palatal gingiva between the left central maxillary incisor and the left lateral maxillary incisor. Applying continuous compression to the area with sterile gauze for over 20 min controlled the bleeding that was very copious from this source. The patient was monitored for 1 h prior to dismissal and was instructed to gently rinse the area with chlorhexidine 0.12% (twice daily for one week) alongside weekly monitoring. A complete blood count was ordered by her oncologist and revealed findings within normal levels with normal cell counts. The platelet count was 366 K/uL and the prothrombin time and activated partial thrombplastin time were all within range. There were no further reported episodes of intraoral bleeding or signs of gingival disease at subsequent follow-up visits at one, two and four weeks. The patient was advised to return for immediate follow-up if additional episodes of intra-oral bleeding occur. |
A 25-year-old female patient was admitted to the emergency service with multiple traumas following a motor vehicle accident. The patient was reported to have been riding in the back seat of the vehicle without the safety belt fastened when the vehicle rolled down an embankment. The patient was conscious but agitated. Bilateral globes were completely out of their sockets (fig. ). During examination, the right pupil was dilated and bilaterally did not react to light. The patient had no light perception bilaterally. The right globe was intact in appearance but hypotonic. The anterior segment of the left globe was normal; however, the right fundus could not be visualized. The left eye fundus examination revealed patches of hemorrhage around the optic disk. The patient had deep cuts of 4 cm towards the right lateral section of the upper lip and 5 cm on the lower jaw.\nA cranial and orbital tomography revealed multiple comminuted fractures of the nasal bones, ethmoidal bone, both zygomatic bones, hard palate, right maxillary alveolar bone, and right pterygoid process. The fracture arch passed through the whole walls of both orbits except for the orbital roofs, all walls of the right maxillary sinus and all walls of the left maxillary sinus except for the anterior wall were classified as Le Fort II fracture. There were also fractures of the bony septum and the sphenoid sinus. All paranasal sinuses were filled with blood. There was a displaced fracture of the right mandibular ramus and angulation of the condyle to the medial aspect. There was a comminuted fracture of the left mandibular fossa resulting from impaction of the condyle.\nThe patient had an intraorbital hematoma in the right eye. There was rupture of all extraocular muscles of both eyes, except for the superior and lateral rectus of the left eye. Bilateral optic nerves were avulsed at the orbital apex and retracted toward the posterior bulbus (fig. ). The right optic nerve was retracted 2.5 cm to the nasal side and the left optic nerve was retracted 2 cm. Brain parenchyma was normal.\nIn addition, the left femoral head was dislocated. The patient underwent closed reduction and derotational casting at the orthopedic clinic. She was instructed by the plastic surgery unit to do jaw-opening and closing exercises for mandibular damage.\nApproximately 10 h after hospital admission, the patient underwent reduction of the globe into the orbit under general anesthesia. In the meantime, the globes were preserved and kept wet using artificial tear gel. Rather than a major surgery, it was planned to perform the reduction of the globe into the orbit because of the existence of bilateral transection in the optic nerves of the patient, visual hopelessness, posterior extraocular muscle rupture and the risk of not finding the ruptured muscles. During the operation, the socket was enlarged with lateral canthotomy, and then the globes were gently repositioned into the orbit with the assistance of an orbital plate and Desmarres lid retractor. The patient underwent bilateral temporary tarsorrhaphy, and a compression pad and bandage were applied. The compression closure was removed on day 3 and the tarsorrhaphy sutures on day 10. There was progressive and unresponsive edema in the right cornea. On the subsequent days, an ulcer developed, accompanied by the shrinkage of the globe on the lower half of the cornea (fig. ). Phthisis bulbi occurred in the right eye at month 3. A prosthesis was fit over the phthisis bulbi (fig. ). An anterior segment examination of the left eye was unremarkable except for pupillary dilation. There was an upward and outward deviation of the left eye. Her eye movements were limited in all directions of gaze. Fundus examination revealed optic atrophy. |
A 63-year-old otherwise healthy man underwent transurethral resection of the prostate in 2014 for benign prostatic hyperplasia. After the procedure, he developed panurethral necrosis with consequent stricture. Two urethroplasties for reconstruction of the bulbar and distal urethra using buccal mucosa grafts were performed in 2016 and 2017 by urology team in another hospital. Two months after the last reconstructive procedure, the stricture relapsed and a dilation was performed, resulting in a satisfactory urinary flow for 6 months. He underwent another urethroplasty using a preputial flap in a different institution and kept urinary catheterization for additional 6 months. After catheter removal, several fistulas in the penile urethra were found. The patient was under various antibiotic regimens and underwent cystostomy. He presented to our emergency service with penile infection, dehiscence of the balanopreputial sulcus, and partial necrosis of the ventral and lateral aspects of the glans, cavernous bodies, and distal penis (\n). Empiric antibiotherapy was administered and later adjusted to the microbiological and antibiotic susceptibility testing results. A magnetic resonance imaging confirmed necrosis of the left aspect of the glans, inflammation of the subcutaneous tissues, and possibly a urinoma. A biopsy ruled out squamous cell carcinoma. On December 9, 2019, he underwent surgical debridement of the unviable tissues and reconstruction using a right ALT flap based on two musculocutaneous perforators, extended laterally to the vastus lateralis muscle to include a segment of fascia lata and elevated as a composite flap (\n). The main pedicle-descending branch of the lateral circumflex femoral artery was dissected until its origin and isolated to preserve the motor nerve to the vastus lateralis muscle and rectus femoris muscle. The entire length of the perforator flap pedicle was 16 cm. The flap was tunnelized under the rectus femoris and a groin subcutaneous tunnel.\nThe extended segment of fascia lata was used for Buck's fascia replacement, as a layer of deep fascia for circumferential reinforcement and to cover the erectile bodies of the penis. The glans was kept in place given its satisfactory perfusion by the end of the procedure, despite near-total amputation.\nIn the postoperative period, the flap showed good perfusion, however, the glans presented with venous congestion and developed necrosis during the following days. After extensive multidisciplinary discussion, the urethra was considered irreparable to allow voiding through the end of the penis. One week later, he was reoperated for surgical debridement of necrotic tissues of the glans, and a perineal urethrostomy was performed by the urology team to create a permanent opening into the urethra through an incision in the skin of the perineum (\n). After the second procedure, the patient had purulent drainage by the scrotum basis: a\nKlebsiella pneumoniae\nsensible to ertapenem, meropenem, and amikacin was isolated and the patient was treated accordingly, with good response to the treatment. Finally, on January 20, 2020, the patient underwent a revisional procedure for flap remodeling. He had a good postoperatory evolution, the urinary diversion (suprapubic cystostomy) was clamped, and the urethral catheter was taken out 3 weeks postreconstruction. After the new opening for urine to pass was created in the perineum, he was able and easily adapted to void in a seated position, maintaining urinary continence. He was discharged on February 14, 2020. After 12 months, there were no signs of recurrence or wound dehiscence. Erectile function was not present until last observation, and a urology appointment was scheduled for planning penile prosthesis insertion. Fat defatting/thinning was unnecessary due to the initial extensive and deep defect. The patient was satisfied with the flap bulk and donor-site scar. A good aesthetic result was seen (\n). |
A ten-year-old girl was admitted to our general hospital with numbness of her left palm and fingers in the last 5 months before admission. At that time, she was hit by a car while she was riding a bicycle. The car was coming from opposite side, and she fell with her left forearm was sliced by licensed plate of the car. There was a semicircular open wound with active bleeding on the left forearm, and she was in pain. She was brought to a nearby clinic and had her left forearm sutured. After the pain subsided, she felt numbness of her left hand and fingers. In addition, she could not extend her fingers. Finally, the patient decided to seek medical attention and get further treatment at our general hospital.\nFrom physical examination, there were claw hand deformity with thenar and hypothenar atrophy as well as a scar on the anterior side of distal forearm (). Sensorium loss of the palm and third, fourth, and fifth fingers was impaired. No tenderness was found. Capillary refill of the fingers was normal. Range of motion of the fingers was altered with limitation of finger abduction and thumb apposition (). Moreover, range of motion of the wrist was within normal limit.\nRoutine laboratory examination was within normal limit. The patient was taken for wrist and forearm radiographs and, similarly, there was no abnormality depicted on either bones or soft tissue.\nThe patient also underwent electromyography examination which showed median and ulnar nerve lesion at the left forearm with total axonal degeneration. No signs of reinnervation of both peripheral nerves were detected.\nThe patient was diagnosed as ulnar and median nerve palsy of left forearm, and then we planned to perform surgical exploration of the nerves and to repair with sural nerve graft, Zancolli procedure and sural nerve graft.\nIntraoperatively, skin incision was made on the previous surgical scar. Injury site was explored, and complete rupture of both ulnar and median nerves was found. Degeneration of both nerves was also seen, with neuroma rising from both the proximal stumps. The proximal and distal ends of both ulnar and median nerves was cut until nerve fascicle was visible. The distance between proximal and distal stump was measured: for ulnar nerve the distance was 7 cm, while it was 8 cm for median nerve. Sixteen centimeters of ipsilateral sural nerve was harvested, and the ulnar and median nerves were repaired using the nerve graft. Then Zancolli procedure was performed: skin incision was made along the palmar crease, A1 pulley was identified around metacarpophalangeal joint, longitudinal incision was made on the pulley, flexor digitorum superficial tendon was retracted laterally, metacarpophalangeal joint capsule was identified, an elliptical incision was made over the joint capsule, and capsulodesis was performed. Postoperatively the wound was closed and immobilized by elastic bandage ().\nWe followed the patient at 3-week postoperatively, and the patient had improvement of her claw hand (). She was advised to continue her rehabilitation of her hand to further improve her hand function, especially opposition and key pinch. At 6-month follow-up, she had improved grip strength and normal functional level of her left hand. At 2-year follow-up, she could handle daily activity as before the accident and was satisfactory with her condition. () |
We report the case of a 77-year-old female with left elbow pain for five months without any prior trauma who received initial treatment at a local hospital in Southern Thailand. She was diagnosed with septic elbow, and open arthrotomy of the elbow was performed and she was given IV antibiotics. The culture results did not identify any specific causative organism at that time. Her symptoms did not resolve, she had a chronic wound and persistent elbow pain, and she was referred to our institution, the major tertiary care and referral center in Southern Thailand, for further investigations and management.\nShe did not have any underlying diseases. The history regarding past pulmonary infection was negative. She did not take any immunosuppressant or herbal medicine. The physical examination at our institute showed a chronic wound at the lateral aspect of the elbow about 0.5 x 2.0 cm in size with clear yellow fluid discharge. The elbow was swollen and warm. Her elbow range of motion was limited due to pain. Laboratory investigations showed a white blood cell count of 4,190/μL with 63% polymorphonuclear neutrophils (PMN). The renal function tests and electrolytes were all within normal limits. Erythrocyte sedimentation rate and C-reactive protein were elevated at 114 mm/hr and 8.48 mg/L, respectively. Radiographs revealed osteolytic lesions around the elbow joint and severe bone destruction with dislocation of the elbow joint (Figures , ).\nA magnetic resonance imaging (MRI) showed abnormal bone marrow edema involving the distal humerus, proximal radius and ulna with a moderate amount of joint fluid with diffusely enhanced thickened synovium. There was severe destruction of the elbow joint (Figures -), which we diagnosed as septic arthritis and osteomyelitis of the elbow.\nDue to the chronic presentation, negative culture results and severe destruction of the joint, we suspected an atypical infection such as TB. We suggested surgery for the definitive diagnosis and as having the best likely outcome, and the patient agreed. Chest radiograph was also reviewed and did not find any evidence of pulmonary TB.\nIn the OR, the elbow was approached posteriorly through a midline longitudinal incision, and necrotic tissue with a cheese-like appearance with no frank pus was found. Copious irrigation and debridement were done.\nA subsequent tissue acid-fast bacillus (AFB) stain was positive with 4 AFB cells/hpf. Polymerase chain reaction (PCR) was also positive for M. tuberculosis, and culture later was positive. Two days later, the patient was brought to the operating room for a second time to repeat the irrigation and debridement of the elbow joint because of persistent fever. The ulnohumeral joint was found to be dislocated and was reduced and stabilized with a pin (Figures , ). The final diagnosis of TB septic arthritis and osteomyelitis of the elbow was confirmed.\nAfter the operation, a regimen of anti-TB drugs was prescribed by an infectious disease specialist, consisting of isoniazid (INH), rifampin (RIF), ethambutol (EMB) and pyrazinamide (PZA) at dosages of 300, 600, 1,000 and 1,000 mg/day, respectively. The regimen was planned for 12 months according to CDC guidelines []. However, early in the treatment, she developed nausea/vomiting with a transaminitis liver profile. Her aspartate aminotransferase (AST) elevated from 21 U/L to 83 U/L and alanine aminotransferase (ALT) elevated from 9 U/L to 31 U/L. The pyrazinamide was considered to be the most likely cause and was changed to levofloxacin 500 mg/day. The liver function test and the symptoms still did not improve, so the regimen was changed to levofloxacin 500 mg/day, streptomycin 600 mg IM three times/week and ethambutol 800 mg/day, following which her symptoms and liver function gradually improved.\nThe wound eventually healed, and the stitches were removed at two weeks' post-operation. The elbow was immobilized in a posterior slab for six weeks, at which time the pin was removed, and the slab replaced with an elbow splint, which allowed a limited extension of 30 degrees. She began a range of motion exercises at eight weeks. At three months' post-operation, she was able to flex her elbow to 110 degrees with an extension lag of 30 degrees. The disabilities of the arm, shoulder and hand (DASH) score improved from 55.8 at presentation to 28.3. At her last follow-up seven months' post-operation, she had mild pain (pain Visual Analog Score = 2-3) with elbow motion with the same arc of motion as at the previous visit. The radiograph shows pseudarthrosis of the elbow (Figures , ). After that, she did not come for any more follow-up visits. |
A 37-year-old woman referred to our clinic with complaints of neck swelling since the past 2 years, which was tender and had infectious secretions after a few months. This problem was first seen only during eating and got slightly better with traditional herbal remedies. After a year, previous treatments did not work, and the patient was annoyed by a permanent pain. In clinical examinations, a solid swelling was observed in the right lateral region of the neck under the jaw angle, and regional lymphadenopathy was evident (Figure ). Suppuration secretion was not clearly visible in the mouth. A stiff and painful swelling was seen in the mouth near the Wharton duct (Figure ). After taking panoramic view, sialadenitis was diagnosed to have occurred due to sialolith in the right submandibular gland (Figure ). Regarding the chronicity of sialadenitis and the history of infectious secretions in the submandibular gland, the selective treatment was removal of the gland by surgery. However, to prevent possible injuries such as damage to the marginal mandibular nerve and skin scar after healing, it was preferred to use a more conservative treatment. The risk of recurrence of the lesion was explained to the patient. Due to the presence of stones above the mylohyoid muscle, it was decided to remove it by intraoral approach. After initial medical evaluations, the patient was placed under general anesthesia and stone was removed through a longitudinal cut along the duct of the salivary glands. Our goal was to cannulation the salivary gland duct, which was done using a pink peripheral venous catheter with an external diameter of 1/1 and an internal diameter of 0/8 (Figure ). First, the catheter was placed into the Wharton duct by a needle. Then, the needle was removed and the catheter remained in the duct. Several back and forth movements were performed in the direction of the duct to remove the obstruction, and the orifice of Wharton was sutured to the floor of the mouth. Narrowing the Wharton orifice would prevent the saliva from escaping, and the obstruction would not come back in the near future. The patient's conditions were satisfactory after a 1-year follow-up (Figure ), and no opacity was seen in the panoramic view (Figure ). |
A 69-year-old Filipino man with history significant for hypertension and hyperlipidemia presented to his primary care physician with hematuria with weight loss of 1 month’s duration. He did not have any flank pain, burning on urination, or increased urinary frequency. He did not endorse any symptoms of fatigue or night sweats. His only medication was atenolol for his hypertension. He did not smoke tobacco, drink alcohol, or do any recreational drugs. He was unemployed at time of interview. He did not have any family history of cancer. His vital signs were within normal limits. On physical examination, he was well appearing and in no acute distress. He had no palpable mass and had an otherwise normal cardiovascular, respiratory, and neurologic examination. Laboratory work showed normal cell counts and normal electrolytes; the results of his kidney and liver function tests were normal. A computed tomography (CT) – intravenous pyelogram was performed as a diagnostic work-up for his hematuria, which demonstrated a large mass in the left collecting system and proximal ureter. He was seen by urology with plans for surgical resection 1 month later. Three weeks later he was admitted to the Emergency Department with nausea and vomiting. He was tachycardic to 110 beats per minute but maintained a normal blood pressure. His laboratory results were notable for hemoglobin to 12.1. His sodium was 134. At that time, a CT scan of his abdomen and pelvis showed interval enlargement of the left renal mass. An ureteroscopy with biopsy was performed, which showed necrotic tissue with rare crushed degenerating atypical cells. A screening chest CT scan was also obtained which showed a small 3 mm nodule in the lower lobe of his left lung. A follow-up interventional radiology-guided left kidney biopsy showed a cellular neoplasm with sheets of pleomorphic round cells with hyperchromatic nuclei, irregular nuclear outlines, and inconspicuous nucleoli with scant and delicate cytoplasm which is consistent with SCC. The tumor cells were positive for the neuroendocrine markers synaptophysin and CD56 with focal staining for chromogranin and dot-like positive staining for cytokeratin (AE1/AE3), supporting the diagnosis of SCC (Fig. ). A bone scan did not show any metastatic lesions. Shortly afterwards, he developed dizziness and an MRI of his brain was obtained revealing a 1.6 cm partially hemorrhagic round mass with surrounding edema in the midline superior vermis potentially representing metastatic disease. An additional 4–5 mm hemorrhagic metastatic focus was seen in the right occipital convexity. The cerebellar mass was resected and probably represented a renal origin due to the absence of lung masses along with clinical and radiographic correlation. He was started on whole brain radiation therapy during his in-patient stay. An out-patient oncology referral was made but he was unable to establish care due to frequent hospitalizations. He had several hospital admissions for nausea and vomiting and continued to decline functionally. He developed chronic hyponatremia during these hospitalizations which were attributed to SIADH. He originally presented with sodium of 119 and was stabilized to a sodium level of 128 with the use of salt tablets. He declined chemotherapy when it was offered by the oncology team during in-patient consultation due to poor quality of life and functional status; he died within 8 months of presentation at his nursing facility. The cause of his death was unknown. An autopsy was not performed. |
A 60-year-old male patient presented to our clinic with a continuous nasal discharge and nasal congestion for years. The patient also had complaints of snoring and problems regarding the senses of smell and taste. His anamnesis revealed that he did not know any information about his birth or childhood. It was learned that he had undergone a nasal operation under local anesthesia about 40 years ago, but he had not had any advantages of that operation and had not applied to another physician. He had no history of head trauma or radiotherapy. In the endoscopic nasal examination of the patient, the nasal septum was seen to be located in the midline, the inferior and middle turbinates were atrophic, and bilateral posterior choanal plates were found to be atretic (). No additional congenital anomaly was found in the patient. In the computed tomography of the paranasal sinuses, osseous choanal atresia was detected on the right posterior nasal cavity and membranous choanal atresia on the left side (). Maxillary, ethmoid, and frontal sinuses were found to be pneumatized whereas sphenoid sinuses were not developed.\nBased on these findings, the patient underwent a transnasal endoscopic surgical treatment under general anesthesia. Endoscopic examination was performed via aspirator palpation and the choanal atresia area in the left nasal cavity was found to be membranous type. The mucosal membrane was perforated with a blade and the curved aspirator passed through the mouth was observed in the nasopharynx. Following the mucosal flap elevation, the surrounding bone tissue was removed using a curette and Kerrison punch forceps and the mucosae were laid toward the nasopharynx. After obtaining an adequate choanal opening on the left side, the right nasal cavity was operated. Choanal atresia on the right nasal cavity was observed to be mixed type. After the mucosal flaps were elevated, the medial pterygoid process and vomer were drilled out. Mucosae were laid down to obtain a choanal opening on the right side as well. For stenting, a six mm endotracheal tube was placed to the right nasal cavity in a way that the balloon would be on the choana and fixed to the membranous septum. No stent was placed on the left side. Nasal irrigation was started after the operation. The nasal stent was removed 3 weeks later and adequate openings were seen in the choanae (). The patient has been followed in our clinic for a year without any problem. |
A psychiatry consult was required for a 41-year-old woman from the Tertiary Ophthalmology Hospital, Tabriz University of Medical Sciences. She was admitted with symptoms of endophthalmitis in the right eye and further evaluations reported the presence of multiple sewing needles in head and neck. Vision of both eyes was intact and no abnormal sign was observed in neurological examinations. After appropriate treatments for endophthalmitis, the patient was transferred to Razi Hospital, the university mental hospital, for further evaluations.\nThe patient was a graduated, married housewife. She had two daughters, who had married and moved out. Her husband was military staff and they have been married for 22 years. The patient was admitted with stable vital signs. She was uncooperative and indifferent about being admitted to a hospital. She had no special complaint except for a mild headache and gave short answer to direct questions about her situation.\nThe most important findings in her mental status were a decreased eye contact, a monotone voice, no spontaneous speaking, paucity of answers, blunt affect, and no insight. She described herself as depressed and tired but denied any delusion or hallucinations. She also did not answer direct questions about the needles. Her family explained that they had noticed a gradual change in her behaviour during the last year or two, beginning with decreased interest in family conversations which were almost her only social relationships as none of their relatives live in her home town. Later they noticed that she had become less interested in cooking, housework, and her own hygiene. She had also complained about intrusive voices coming from neighbours a few times, but her family did not recognize the reason or the importance of such complaints. Persistence of these problems motivated the family to see a general physician. A diagnosis of depression was made based on her symptoms and the doctor prescribed antidepressant medications to help her with sleep problems. She could not take the pills regularly and her symptoms continued, while her daughter noticed the red eye and took her to the hospital.\nPast medical history and family history were unremarkable. The patient's family described her premorbid personality to be more or less shy but eager to communicate with relatives, introverted, and tolerant. No information was available about her developmental history. She got married in a traditional way, in which a premarital relationship is not encouraged. She seldom complained of marital conflict but her relationship with her husband was very limited because of his busy life.\nX-ray anteroposterior and lateral views showed the presence of multiple sewing needles in the head and neck (). Besides the described findings in her eye, several indurations resembling an old self-infliction were palpable in her neck, while the skin was intact.\nBased on the described information, a preliminary diagnosis of psychotic disorder not otherwise specified was made. Risperidone was initiated with 1 mg and increased to 6 mg within two weeks, along with daily visits. Following correction of her sleep pattern, the patient started to cooperate. She could explain that she could hear the voices of neighbours arguing about her and sometimes about her daughters. She did not plan to do something in return as she was sure it would not work; the neighbours talked about her just after her marriage when she moved to the military facility with her husband. They questioned her loyalty to her husband but stopped shortly after she gave birth to her first daughter. She could not talk about these issues with her husband because he had already warned her not to get involved with neighbours.\nShe began hearing these voices after the marriage of her youngest daughter, when she felt lonesome a few days after the ceremony. This time, she could hear their voices clearly, even in the hospital, but they were more frequent at home where she was alone most of the day. The humiliating voices never commanded her but made her very upset. She did not feel like doing anything as she felt a lot of pressure. She had no explanations for not taking the prescribed medications, and it was considered as a manifestation of avolition. The only way to get rid of this pain was to inflict pain on her body using needles. This could stop voices for days. She did not offer any special reasons for inflicting damage to her eye using needles.\nThe diagnosis of schizophrenia undifferentiated was established. The treatment regimen included 6 mg risperidone daily and 2 mg biperiden because of neuroleptic-induced Parkinsonism, which resolved within two days. Supportive psychotherapy was provided to the patient during which she mostly felt like talking about her past. During these sessions, the patient was allowed to talk about her concerns and was encouraged and assisted with every-day life problems and the improvement of her quality of life. Admission continued for two more weeks and her behaviour was closely monitored. She reached a satisfying level of self-care and no self-mutilation attempts were observed. After psychoeducation for the family, the patient was discharged. The psychoeducation included information about the disorder, treatment, and relapse prevention. She could not attain the planned psychotherapy sessions but was taking medications regularly in a one-month follow-up and no positive symptoms of schizophrenia were reported. |
The patient is a 76-year-old woman with a two-year history of left elbow pain empirically diagnosed as gout. When her symptoms failed to improve with appropriate management, radiographs were obtained, demonstrating a lesion in the proximal radius () characterized as a mildly expansile lucent lesion with a thin zone of transition but no sclerotic rim. Internal osseous septations were present and there was cortical thinning but no visible cortical breakthrough, periosteal reaction, calcified matrix, or soft tissue mass. The initial differential diagnosis included metastasis, multiple myeloma, and other less common entities such as a primary sarcoma of bone or atypical infectious process. She was referred to our tertiary care hospital to consult with an oncologic orthopedic surgeon. Further history obtained at that clinic visit elicited that 3 years previously she had incidentally discovered lytic lesions in her skull and left clavicle that were evaluated in another medical system. Biopsy of both lesions performed at that time was inconclusive showing a mix of inflammatory and fibrous cells per report. The pathologic specimens were not available for further review. Physical exam at her clinical visit was unremarkable with no palpable lymphadenopathy and no visible abnormality at the symptomatic left elbow. SPEP and UPEP tests were negative.\nHer initial imaging work-up included CT of the chest, abdomen, and pelvis; contrast-enhanced MRI of the left forearm; and nuclear medicine bone scan. Her CT scan showed no findings of primary malignancy and—pertinent to her eventual diagnosis—showed no lymphadenopathy or vital organ abnormality. Bone scan demonstrated marked radiotracer uptake at the site of the lytic lesion in the proximal left radius as well as at the previously biopsied skull and left clavicle lesions (). The MR scan of the left forearm showed a marrow replacing lesion within the proximal diaphysis of the radius (). The lesion was T1 isointense, T2 hyperintense and demonstrated avid enhancement. Cortical thinning and small areas of cortical breakthrough not visible on the radiographs were apparent on the MRI. No associated soft tissue mass or perilesional edema was present.\nAt the request of the orthopedic oncologist, a fluoroscopy-guided percutaneous biopsy was performed by Musculoskeletal Interventional Radiology. This rendered only tiny fragments of tissue that were nondiagnostic at histologic review. The patient then underwent open biopsy and curettage of the lesion with Orthopedic Surgery for both diagnostic and treatment purposes. Lesion histology demonstrated features diagnostic of RDD including emperipolesis (engulfment of intact lymphocytes contained with the cytoplasm of histiocyte cells) and positive S100 immunohistochemical staining (). At her follow-up clinic visit 8 weeks after surgery, the patient reported resolution of her left elbow pain, and repeat radiographs demonstrated partial filling in of the lesion with healing bone (). She was discharged from clinic and instructed to follow up if she developed recurrent left elbow symptoms or similar symptoms at a new site. One year later, she has not sought further care at our institution. |
Case 2: A 23 year old man attended our clinic requesting a genetic test for HD. His mother developed clinical manifestations of HD in her 30s and was known to our Service. At the time of consultation this man was an inpatient in a mental health facility. He was admitted following a second episode of psychosis, with auditory hallucinations and some atypical features (a body dysmorphic delusion). Motor examination was normal except for mildly impaired eye movements. He was on a moderate dose of atypical anti-psychotic medication but his neurologic examination was reported to be normal prior to initiating medication. He was somewhat reluctant during neuropsychological examination and performed poorly in a range of measures, over a broader range of cognitive domains than one would expect for a typical, early, HD patient. (Fig.3)\nDiscussion: It is possible that this man’s psychosis was part of HD, although his cognitive impairment did not lend as much support to the diagnosis of clinical HD as it would have, if the pattern had been typical for HD. This illustrates some of the difficulties in incorporating psychiatric symptoms into the diagnosis of clinical HD. Since these psychotic episodes occurred in his early twenties, they could potentially be due to schizophrenia rather than HD. If the first psychotic episode had occurred in his early 50s instead, then it would have been a little more specific for HD. This case also demonstrates the flow-on effect of the clinical diagnostic criteria, on the decision to carry out genetic testing on a pre-symptomatic basis or not. Given the uncertainty in his diagnosis, we suggested genetic testing on a pre-symptomatic, rather than diagnostic basis. The geneticist advised him to reconsider the decision to test, given his cognitive impairment. This man has an identical twin who was uncontactable at the time, which added to our hesitation to offer testing. When seen twelve months later, his psychosis had resolved, cognitive performance improved and he no longer wanted to have a genetic test. |
A 9-year-old boy referred to our clinic with complaint of unaesthetic appearance of his upper anterior teeth. The patient had a nonsignificant medical history and no case of fusion was reported in his family. Intraoral examination revealed localized macrodontia present in the maxillary anterior region. Clinically maxillary left central and lateral incisors were found to be fused and indentation running from incisal edge to cervical margin was also observed. Maxillary left central and right lateral incisor crowns were distally tipped. Distolabial rotation of left lateral incisors was present. The labial and palatal aspects of both 21 and 22 teeth were found to be caries free and healthy periodontium. Spacing was present in the maxillary anterior region and distal to lateral incisor in the lower arch ().\nOrthopantogram radiograph revealed incomplete fusion of 21 and 22 at crown level with separate pulp chambers and two distinct roots. Radiographic evaluation also revealed an interference in eruption of permanent canine 23 by distally tipped root of fused lateral incisor (22) (). Treatment was recommended in order to improve esthetic status of the patient, guide the canine into normal eruption path, and intercept developing malocclusion, which may require comprehensive orthodontic treatment in future.\nThe treatment plan was explained to his family and with their consent; the periodontal envelope flap was raised after anaesthetizing right side of the maxillary anterior region. Initially the fused teeth were separated slightly beyond cementoenamel junction using long, thin diamond bur. After that, an elevator was used to separate the fused teeth; successful separation of 21 and 22 was confirmed by clinical mobility of individual tooth and assessed through radiograph. The periodontal flap was then replaced and suture placed.\nAfter a week period, suture was removed and orthodontic treatment with 2 × 4 fixed appliance was begun in maxillary arch. Roth 0.022 slot brackets were bonded on maxillary incisors and preformed molar bands with buccal tube were cemented in 16 and 26. Initially, 0.014 nickel titanium archwire with protective sleeve was used for alignment and leveling (), after that progressively archwires were changed to 0.016 nickel titanium and 0.018 stainless steel wire. Maxillary anterior spaces were closed with elastomeric chain in 0.018 stainless steel wire. Fused teeth were aligned at the end of six-month orthodontic treatment (). Intraoral periapical radiograph showed improvement in root parallelism of 21 and 22 during fixed appliance treatment (). |
A 64-year-old woman visited our orthopaedic department for pain in both knees.
The patient had suffered from pain in both knees for six years and the patient was managed with medications for pain control at a local medical centre. However, the pain was more aggravated from six months prior, and the medication was not sufficient to control the pain. After the patient had visited our orthopaedic department, the patient was diagnosed as having osteoarthritis in both knees and total knee replacement was recommended ().
The patient was relatively healthy without any previous medical or surgical history except for hypertension. The patient had a limitation of motion in both knee joints, but there were neither external wounds nor sensory changes in both lower extremities.
Preoperative examinations such as routine laboratory tests and imaging studies such as chest X-rays and a pulmonary function test were within normal limits. The patient underwent total knee replacement surgery in both knee joints ().
On the second postoperative day, the patient demonstrated stuporous mentality with eyeball deviation and became markedly dyspnoeic and hypoxemic. Arterial blood gas analysis showed both a decreased PO2 as 33.9 mmHg and an oxygen saturation level of 68.4%. Brain MR and pulmonary CT imaging were performed to exclude cerebral and pulmonary fat embolism. Brain MRI demonstrated multiple small dot-like high signal intensities in both the centrum semiovale and subcortical white matter on diffusion-weighted imaging (DWI), which was suggestive of cerebral fat embolism ().
High resolution CT of the lung demonstrated the presence of patchy ground glass opacity in both lungs and bilateral pleural effusion. An enhanced chest CT image demonstrated the presence of a small embolus in the right main pulmonary artery. The mean attenuation value of the embolus was -42 Hounsfield units (HU), suggestive of fat embolism ().
The patient was treated supportively with mechanical ventilation, supplemental oxygen and heparinization. The patient showed gradual improvement of the respiratory and neurological status and no further complications were noted such as |
A 44-year-old male with the past medical history of essential hypertension and tuberculosis presented to the emergency room (ER) with the complaints of high grade fever, fatigue, and shortness of breath. According to patient, he developed high grade fever 1 week ago that was accompanied by myalgias, frontal headache, and generalized weakness. Fever was followed by shortness of breath and exercise intolerance which started 3 days ago and worsened with time. History was positive for mild paroxysmal nocturnal dyspnea and orthopnea though he denied any chest pain, palpitations, cough, or syncope. He also had frequent light headedness and got 1 episode of coffee ground emesis after which he was brought to ER. There was no history of blood in stools or urine. The patient denied any history of diabetes, ischemic heart disease, or hyperlipidemia. The patient was only taking HCTZ for hypertension and did not have any allergies. He was an active smoker with a history of one pack of cigarettes daily but denied use of alcohol or illicit drugs. The family history was positive for myocardial infarction in father at the age of 77. The patient did have a history of recent sick contact with one of his brothers who had dengue fever 3 weeks ago.\nReview of patient's past medical records indicated that patient had dengue fever 1 year ago for which he was admitted on the floor but fever got resolved on its own with no complications. At that time patient developed vague chest discomfort for which EKG was done which showed widespread T wave inversions. Cardiac enzymes were mildly elevated and echocardiography showed normal wall thickness and ejection fraction of 58%. Taking into account above findings, a tentative diagnosis of mild dengue myocarditis was made. CXR was normal and patient was discharged. Endomyocardial biopsy and cardiac MRI were not done as the patient had mild myocarditis with normal myocardial functions. Follow-up CXR, Echo, and EKGs were normal and patient remained healthy other than having slight fatigue.\nIn ER patient was lethargic and seemed anxious due to generalised body aches and headache. On physical examination, vital signs were as follows: blood pressure of 89/52 mmHg, temperature of 103 F, heart rate of 116/min, and respiratory rate of 19. Chest examination revealed reduced breath sounds at lung bases bilaterally with scattered fine crackles in both lungs; no rhonchi or wheeze was heard. Examination of heart was remarkable for normal S1 and S2 with no murmur or clicks; an S3 gallop sound was audible. There was no pallor, scleral icterus, cyanosis, clubbing, or peripheral edema. The rest of the physical examination was unremarkable.\nPatient was suspected to have dengue shock syndrome and was immediately admitted on floor and resuscitated with IV fluids. Antiemetics and Tylenol was also given. Routine EKG was done which showed sinus rhythm and frequent premature ventricular contractions (PVC) with no ST or T wave ischemic changes (). CXR showed bilateral interstitial edema, cardiomegaly, and pleural effusion in both lungs (). The X-ray findings were suggestive of acute congestive heart failure. Elevated BNP (1040) supported the X-ray findings His haematological investigations revealed slight thrombocytopenia (127000), leucopenia (3100), and haemoglobin of 12. Cardiac enzymes were initially slightly elevated but remained stable over the course of hospital stay (). Urgent transthoracic echocardiography was done which showed ejection fraction of 10% and left ventricle dilation without focal wall motion defect and or focal thinning (). Cardiac catheterization revealed EF of 13% and normal coronary arteries (). Patient was started on dobutamine infusion along with ACEIs, spironolactone, and Lasix. Blood cultures, ESR, hepatitis panel, thyroid profile, and urine drug screen were all within normal limits. Malarial antigen test and typhoid serology were also negative. Anti-dengue IGM by ELISA was negative at the time of admission but was positive for IGG antibodies at that time. Repeat testing on day 3 showed positive IGM antibodies for dengue. The diagnosis was confirmed with reverse transcriptase polymerised chain reaction (RT-PCR) which was positive for dengue virus serotype 3 (DEN-3). In view of above clinical scenario with positive dengue serology, history of myocarditis during primary dengue infection 1 year ago, and echocardiographic findings, diagnosis of dengue virus induced dilated cardiomyopathy (DCM) was made. Fulminant myocarditis with acute myocardial failure was ruled out as cardiac enzymes were almost normal in the setting of dilated cardiac chambers on Echo.\nPatient condition worsened gradually and he developed acute hypoxic respiratory failure for which he was intubated on day 4. Patent developed severe thrombocytopenia and despite platelet transfusions he developed massive lower GI bleed leading to refractory shock. Patient was transfused 2 units of PRBC and platelets, but despite aggressive resuscitation and intensive care, patient died on day 5. |
A 33-year-old gravida 3 para 1021 (one term pregnancy, no preterm pregnancies, one ectopic pregnancy and one spontaneous miscarriage, and one living child) Hispanic woman with a history of a previously diagnosed cornual ectopic pregnancy in a unicornuate uterus presented for evaluation and treatment of suspected persistent POC in the left cornu of a unicornuate uterus. Her pregnancy history is notable for a term uncomplicated vaginal delivery and prior early first trimester miscarriage managed with expectant management. She had no other significant medical or surgical history. She was diagnosed with a left interstitial pregnancy and treated with systemic methotrexate, receiving a total of four doses. Given her desire to conceive again, she underwent a gynecologic ultrasound, which revealed a persistent gestational sac and fetal pole in the left cornu despite multiple confirmatory hCG values <5. She was asymptomatic but was referred to the reproductive endocrinology service for management of this residual mass given her desire to try to conceive again and the potential to utilize assisted reproductive technology in future cycles. Ultrasound revealed a gestational sac in the left cornu measuring 10×7 mm with generalized reactive muscular echogenicity surrounding the sac and a fetal pole measuring 6.2 mm (Fig. ). Ultrasound findings were suggestive that these persistent POC would be accessible via suction D&C based on its continuity with the endometrial stripe on ultrasound.\nShe was counseled regarding the following options: further expectant management with serial ultrasound monitoring given no known evidence that persistent POC can result in uterine rupture, and surgery. She elected to proceed with definitive surgical treatment given her desire to conceive and the potential catastrophic nature of uterine rupture in her unicornuate uterus. Intraoperative transvaginal and transabdominal ultrasound again confirmed persistent left interstitial pregnancy with POC within surrounding decidualized endometrium. A family planning specialist was consulted during this case. First, suction D&C was attempted. A 7 mm flexible cannula was inserted to the fundus under transvaginal ultrasound guidance. The left cornu was unable to be accessed despite multiple attempts with transabdominal and transvaginal ultrasound guidance. This was also attempted with a 7 mm rigid curved cannula but similarly was unsuccessful.\nHysteroscopy was then performed using a 5 mm Karl Storz operative hysteroscope with a 2.9 mm 30° lens, 5mm sheath, and 5-Fr operating port with normal saline as the distending media. Upon entry of the hysteroscope into her uterus, tubal ostium was identified. However, no gross POC were visualized within her uterine cavity. Next, a Novy™ Cornual Cannulation Set (Cook Medical) was passed hysteroscopically into the left cornual region under hysteroscopic and ultrasound guidance (Fig. ). Although the device was successfully cannulated into the left cornu, no tissue was able to be aspirated (Fig. ). As an alternative intended to grasp and remove the POC, a Tricep™ extra-strength hooked-prong grasping forceps (Boston Scientific) with a 3.0-Fr sheath and 120-cm working length (urologic stone retrieval basket) was suggested based on prior cornual procedures with a similar device []. This device was placed into the left cornu under both hysteroscopic and ultrasound guidance (Fig. ); it was opened and closed within the cornual region several times. Dense fibrous tissue was grasped and removed with visible POC. This was repeated several times in a similar fashion. Karl Storz 5-Fr hysteroscopic grasping forceps were also used to grasp tissue extruding from this cornu several times (Fig. ). At the end of the case, ultrasound revealed resolution of the myometrial decidual reaction with removal of persistent POC (Fig. ). Safety was assured during the case with constant hysteroscopic visualization; transabdominal ultrasonography was also utilized to assure excellent visualization at all times. Laparoscopy was not necessary in this case due to combined hysteroscopic and ultrasonographic visualization during the case. The patient was counseled that the resolution of the persistent POC on ultrasound and hysteroscopy decreased the risk of uterine rupture but she was cautioned regarding the continued potential for rupture. Pathology revealed fragments of necrotic chorionic villi and decidua. She was discharged home the same day and had an uncomplicated postoperative course. Postoperative beta hCG was measured again postoperatively and was <5 mIU/mL. |
An 83-year-old man presented to the prosthodontic specialty clinic to enhance the existing maxillary complete denture and/or to make a new denture with or without implant utilization. The patient was referred to prosthodontic clinic by an undergraduate student because of the patient's continual complaint about poor retention and stability of the maxillary complete denture. The patient was healthy and independent with hypertension that was medically controlled. The existing prosthesis, which was fabricated by the undergraduate student one year earlier, included a maxillary complete denture and a mandibular transitional RPD Kennedy Class I modification 1 which were made after extraction of his mandibular incisors keeping only the two mandibular canines and the first left premolar. The patient was not happy with the existing prosthesis and was not totally satisfied with a previous denture made by his dentist four years earlier. His major complaint with the first and second prostheses was mainly that the maxillary complete denture loosens while he is speaking. Multiple chair side hard and soft relines were performed over the previous year to enhance the retention and stability of the existing maxillary complete denture. Because of patient financial limitations, he wanted to avoid implant therapy if a new denture could be fabricated with adequate retention and resistance.\nUpon intraoral examination of the maxilla, the maxillary right and left buccal frena appeared as thick/multiple fibrous bands giving them a "fan-shape" and along with the labial frenum, they were considered low and located close to the crest of the residual ridge (). The vestibules were considered shallow because of the frena anatomy; otherwise, the rest of the soft tissues appeared within normal limits. The edentulous ridge appeared broad, rounded, and covered by firm soft tissue. The patient was informed about the observed frena and had been told that they could be a causative factor for his complaint and for maxillary denture looseness. Therefore, he was advised that the frena should be removed surgically by a procedure called "frenectomy" before a new denture was made. Additionally, he was told that if low satisfaction with the new complete denture continued, implant therapy would be considered. Before the patient was referred to the oral surgeon, the existing soft liners on the maxillary complete denture were removed and a new chair side hard reline (Flexacryl Hard, Lang Dental Mfg. Co, Inc., Wheeling, IL, USA) was performed. shows the significant amount of relief (labial and buccal notches) needed to accommodate the frena after the chair side reline procedure. After the hard reline, clinical evaluation of occlusion and vertical dimension for the existing removable prosthesis found to be adequate. However, during subsequent visits patient was still complaining about the maxillary denture looseness at different time of day while he was speaking.\nThen the patient was referred to the oral surgeon to perform the frenectomy. At the time of appointment for frenectomy, the treating prosthodontist was available to utilize the existing denture as a stent to support and stabilize the tissue attachments in the new position. Because of the existence of broad frena and shallow vestibules, the oral surgeon decided to perform the frenectomy with the Z-plasty technique because of its usefulness in such situation where simultaneous frenum elimination and vestibule lengthening can be achieved. When simultaneous frena excision and vestibule deepening with the Z-plasty technique were completed, and after final suturing, a soft denture reline material (Coe-Comfort, GC America, Inc., Alsip, IL, USA) was placed on the existing denture and carefully inserted into the patient's mouth. A gentle border molding was performed before the denture was removed from the mouth and then the excess reline material was trimmed around the borders. According to a previously suggested protocol,, the patient was instructed to keep the denture in place for the next 24 hours including sleep time and to minimize the in-out placement of the denture during the initial stages of healing (normally the first 3 - 5 days). The sutures were removed after 7 days. During post-surgical week one and two, the initial soft denture reline material was replaced with new material twice. On the third post-surgical week, a different denture reline material was used (Coe-Soft, GC America, Inc.) and left in place for an additional three weeks., The definitive maxillary complete denture construction was initiated six weeks following the surgical frenectomy procedure.,,\nThe full mouth rehabilitation of the patient was achieved following the proposed treatment plan and the rendered treatment was performed according to classical recommendations in text books and literature, for delivering such prosthodontic therapy. The final complete maxillary denture had longer border extensions ( and ). Post-treatment follow-up appointments indicated that the patient was fully satisfied with the final treatment outcome. Currently, it has been over a year since the treatment was completed and the patient is functioning well with the denture. |
A 60-year-old male patient was referred to our institution under the diagnosis of perihilar cholangiocarcinoma. Computed tomography (CT) findings were compatible to those of hilar cholangiocarcinoma Bismuth-Corlette type IIIa with encasement of the right hepatic artery (). A percutaneous transhepatic biliary drainage (PTBD) tube was inserted into the right hepatic duct, and crossed over the hilar stenotic portion and then passed into the ampulla of Vater for probably simultaneous internal drainage. Since we were not accustomed to such placement of PTBD tube and total bilirubin level decreased gradually like in the usual perihilar cholangiocarcinoma cases, we did not perform any other procedure for biliary drainage.\nIn order to induce regeneration of the future remnant left liver, we performed right portal vein embolization (). Two days after this procedure, the patient suffered from paralytic ileus with marked abdominal distension (). Emergency CT scan revealed that marked fluid collection around the liver and whole abdomen, thus percutaneous drainage was performed with insertion of a pigtail catheter over the left liver. The pigtail drainage output was bilious in nature, thus leading the diagnosis of intrahepatic bile duct rupture. Follow-up CT scan showed residual fluid collection in the other area of the abdomen, thus another pigtail catheter was inserted into the pelvis. At this time, we recognized that the underlying cause of intrahepatic bile duct rupture was ineffective drainage of the left hepatic duct with overproduction of bile after right portal vein embolization. Therefore, a PTBD tube was inserted into the left liver ().\nTo enhance the safety of right hepatectomy, we performed additional right hepatic vein embolization to facilitate left liver regeneration ().\nDuring laparotomy, we found that the ruptured area was the diaphragmatic side of the segment II because the superficial liver parenchyma at that area was definitely torn out. After confirmation of respectability with limited dissection of the hepatoduodenal ligament, we carried out routine surgical procedures for right hepatectomy, caudate lobectomy and bile duct resection and reconstruction. Intraabdominal biloma debris and abscess pockets were removed. Multiple abdominal drains were inserted to evacuate residual fluid in the abdomen and pelvis. The patient recovered uneventfully and discharged 18 days after surgery ().\nPathological reports showed that the tumor was a 2.1 cm-sized cholangiocarcinoma with extension to perifibromuscular connective tissue and positive lymphovascular invasion (). There was no lymph node metastasis. No adjuvant treatment was carried out. |
A 53-year-old man presented with a sudden severe headache and drowsy mentality. An immediate CT scan revealed a diffuse, thick subarachnoid hemorrhage in the basal cisterns. In a subsequent DSA, a very small aneurysmal lesion was noticed in the lateral wall of the distal ICA close to the AChA origin. As it had a small, hemispherical contour and located superior to the AChA origin, rather than distal to it, a BBA was suspicious (). Also, associated focal dilatation of the ICA around the hemispherical bulge increased the diagnostic suspicion of a BBA. As good collateral circulation was shown through the anterior and posterior communicating arteries, the crucial surgical goal was preserving the patency of an AChA in addition to close the rupture point of an ICA.\nUsing a left pterional craniotomy, the affected ICA was exposed by opening the carotid cistern and proximal sylvian fissure. The superior half of the circumference of the distal ICA appeared abnormal. The lateral wall of the distal ICA was covered by a blood clot, suggesting a rupture point, while the superior and medial walls were very thin and friable (). Slight manipulation of the medial wall of the ICA to obtain proximal control of the posterior communicating artery (PCoA) via an opticocarotid triangle led to iatrogenic rupture of the superomedial wall of the ICA. Thus, the rupture point was temporarily closed using a curved aneurysm clip, and then attention was paid to the primary rupture point in the lateral wall. After temporary clipping of the proximal ICA and PCoA, the blood clot covering the rupture point was cleared to demarcate the arterial defect and the adjacent AChA. The arterial defect was very close and just superior to the AChA origin. An attempt to suture the defect was failed due to the friable superior wall that was torn. When the curved aneurysm clip was removed, a large round arterial defect was finally revealed (). At this point, a Yasargil aneurysm clip with C-shaped blades (Aesculap Instruments Corp., Tuttlingen, Germany), model FT 824T was applied parallel to the ICA to close the large, round arterial defect, yet this also failed to stop the leakage despite severe luminal stenosis of the ICA. Therefore, the large arterial defect of the ICA was approximated using four stitches with an 8-0 monofilament, resulting in an hourglass appearance due to severe stenosis (). The suture line was then reinforced using the Yasargil aneurysm clip with C-shaped blades, thereby preserving the AChA (). Intraoperative Doppler was used to ascertain the patency of the AChA, PCoA, and ICA.\nThe patient recovered without neurological deficits and returned to his previous life. The postoperative and 6-month follow-up angiograms revealed severely stenotic distal ICA, excellent patency of the PCoA and AChA, and no recurrence of the BBA (), along with good collateral circulation through the anterior communicating artery to the middle cerebral artery. |
A 69-year-old Filipino man with history significant for hypertension and hyperlipidemia presented to his primary care physician with hematuria with weight loss of 1 month’s duration. He did not have any flank pain, burning on urination, or increased urinary frequency. He did not endorse any symptoms of fatigue or night sweats. His only medication was atenolol for his hypertension. He did not smoke tobacco, drink alcohol, or do any recreational drugs. He was unemployed at time of interview. He did not have any family history of cancer. His vital signs were within normal limits. On physical examination, he was well appearing and in no acute distress. He had no palpable mass and had an otherwise normal cardiovascular, respiratory, and neurologic examination. Laboratory work showed normal cell counts and normal electrolytes; the results of his kidney and liver function tests were normal. A computed tomography (CT) – intravenous pyelogram was performed as a diagnostic work-up for his hematuria, which demonstrated a large mass in the left collecting system and proximal ureter. He was seen by urology with plans for surgical resection 1 month later. Three weeks later he was admitted to the Emergency Department with nausea and vomiting. He was tachycardic to 110 beats per minute but maintained a normal blood pressure. His laboratory results were notable for hemoglobin to 12.1. His sodium was 134. At that time, a CT scan of his abdomen and pelvis showed interval enlargement of the left renal mass. An ureteroscopy with biopsy was performed, which showed necrotic tissue with rare crushed degenerating atypical cells. A screening chest CT scan was also obtained which showed a small 3 mm nodule in the lower lobe of his left lung. A follow-up interventional radiology-guided left kidney biopsy showed a cellular neoplasm with sheets of pleomorphic round cells with hyperchromatic nuclei, irregular nuclear outlines, and inconspicuous nucleoli with scant and delicate cytoplasm which is consistent with SCC. The tumor cells were positive for the neuroendocrine markers synaptophysin and CD56 with focal staining for chromogranin and dot-like positive staining for cytokeratin (AE1/AE3), supporting the diagnosis of SCC (Fig. ). A bone scan did not show any metastatic lesions. Shortly afterwards, he developed dizziness and an MRI of his brain was obtained revealing a 1.6 cm partially hemorrhagic round mass with surrounding edema in the midline superior vermis potentially representing metastatic disease. An additional 4–5 mm hemorrhagic metastatic focus was seen in the right occipital convexity. The cerebellar mass was resected and probably represented a renal origin due to the absence of lung masses along with clinical and radiographic correlation. He was started on whole brain radiation therapy during his in-patient stay. An out-patient oncology referral was made but he was unable to establish care due to frequent hospitalizations. He had several hospital admissions for nausea and vomiting and continued to decline functionally. He developed chronic hyponatremia during these hospitalizations which were attributed to SIADH. He originally presented with sodium of 119 and was stabilized to a sodium level of 128 with the use of salt tablets. He declined chemotherapy when it was offered by the oncology team during in-patient consultation due to poor quality of life and functional status; he died within 8 months of presentation at his nursing facility. The cause of his death was unknown. An autopsy was not performed. |
The patient was a 49-year-old Caucasian male with body mass index of 27 kg/m2. He complained about pain on his right knee in the last 8 months. The pain was gradually increased and still could be relieved by rest. He had the history of fixed lateral patellar dislocation since 25 years before. The dislocation happened after he got slipped and his right knee hit the floor with the body on top/kneeling. He came into the examination room with antalgic gait without any walking aid. He felt that his right knee was giving away while walking. There was no other trauma. There were no history of medical and surgical treatment, no family with the same symptom, and no routine sport activity.\nMuscle atrophy could be seen on the anterior compartment of the thigh, specifically on the lateral medial and intermedius muscle. We observed the atrophy of the thigh muscle from the anterior, lateral, and posterior view (). We found the patella was laterally dislocated. The knee range of motion was 0-120° in both passive and active movements. The patellar apprehension test was positive while the other special tests such as anterior and posterior drawer test, patellar tap test, joint line tenderness test, and Lachman test were all negative.\nOn the AP view of the radiographic imaging, we found neither varus nor valgus deformity of the knee and no sclerosis at the subchondral bone. However, whole mechanical axis radiology was not possible to be performed at the time of examination; therefore, we are lacking data such as femoral anteversion and Q angle. There was no sign of secondary osteoarthritis of the knee joint. From the lateral view, there was no sign of patella alta and patella baja, but on the skyline view, we could see that there was a lateral dislocation of the knee. The trochlear groove was relatively normal (). From the physical and X-ray examination, the patient was diagnosed as having chronically fixed lateral patella dislocation of the right knee.\nWe decided to perform extensive lateral release and right medial patellofemoral ligament reconstruction for this case. We performed incision on the lateral side of the knee joint until we reached the lateral capsule. We decided to release the lateral capsule and try to reduce the patella to be in the midline position. The patella was laterally fixed because of the tightened lateral vastus. We extended the incision and performed lateral release by cutting the fascia of the lateral vastus. After that, we tried again to reduce the patella but there was still resistance. We then cut the fascia of the medial vastus until the dislocated patella was fully released (). After we successfully reduced the patella, we performed semitendinosus tendon graft harvesting.\nA 3 cm oblique skin incision was made at the medial proximal tibial area, and the soft tissue was released. The semitendinosus tendon was harvested using a tendon stripper. We made a double-stranded graft by folding the semitendinosus tendon. The graft length was 160 mm, and the diameter of the ST tendon was 7 mm. After preparing the graft, 3 cm longitudinal skin incisions were made at the patellar and femoral natural footprints of the MPFL, and the soft tissue was peeled off so that the graft could pass through the soft tissue tunnel smoothly.\nA patellar tunnel was made at the one-third of the superior part of the patella from the medial side until it reached the lateral side. Subsequently, the graft was passed through the bone tunnel of the patella. After the endobutton (Smith & Nephew, Andover, MA) reached the lateral side, it was flipped to fixate the graft end.\nA femoral bone tunnel was constructed at the MPFL anatomical footprint, which is located at the Schottle point []. A 2.4 mm guide wire was inserted in the medial, ventral, and slightly proximal direction to avoid vessel and nerve injury. A 7 mm drill was used to create a bone tunnel. The depth of the bone tunnel was 80 mm until it reached the lateral cortex of the femur. We then sutured the graft end with ETHIBOND (Ethicon, Somerville, NJ) no. 2 and passed the graft through a medial side of the femur until the end of the tendon coming out from the lateral femoral side ().\nWe then drilled the lateral side of the distal femur and put a cancellous screw and washer with a diameter of 4.5 and a length of 65 mm just above the femoral tunnel. The end of the graft was sutured with ETHIBOND, and the ETHIBOND suture was then anchored to the cancellous screw and washer as the fixation of the patella to prevent it from redislocation ().\nThe soft tissue was then closed in layers. After that, we did confirmation X-ray to confirm the position of the inserted screw at the lateral side of the distal femur (). For the rehabilitation program, at first, the knee brace was applied to keep the knee in a full-extension position. The brace was used in this position up to three weeks; then, the knee flexion was increased to 20 degrees every week. After eight weeks postsurgery, the brace could be discharged and the patient could start active motion of the knee. |
We present the case of a 43-year-old woman, with a known history of classic migraine for the last 20 years, who presented to the ED with a severe headache. Her pain started four days prior to her presentation and progressively worsened in severity. She described the headache as persistent generalized pain, compared with the pulsatile nature of her previous migraine headaches. The headache was worse on awakening and bending. Unlike her usual episodes of migraine, the headache did not improve with the use of oral sumatriptan. She scored the pain as eight out of 10 in severity and it was awakening the patient from sleep. The headache was associated with nausea and recurrent episodes of vomiting. There was no history of preceding head trauma. The patient did not report any history of photophobia, fever, weakness, or loss of consciousness. The patient visited several outpatient clinics who managed her as having tension headache and offered symptomatic treatment only.\nShe reported that the current episode of headache was different in character and severe than her usual episodes of migraine headaches. Of note, the patient was using sumatriptan for the last eight years. Her past medical history is not significant for any health condition other than migraine. She has not undergone any surgical operation in the past. Her family history is significant for classic migraines in her mother and sister. She is a non-smoker and does not drink alcohol.\nUpon examination, the patient looked in pain and distress. She was drowsy but was fully oriented. Her vital signs on presentation were as follows: pulse rate of 114 bpm, blood pressure of 132/78 mmHg, respiratory rate of 18 bpm, temperature of 36.7℃, and oxygen saturation of 99% on room air. Neurological examination revealed grossly intact cranial nerves, no focal motor or sensory deficits, and normal cerebellar examinations. Funduscopic examination showed normal findings. The cardiorespiratory examination was unremarkable. There was no neck stiffness and the meningeal signs were not present. Her laboratory findings revealed a hemoglobin level of 13.9 g/dL, a white blood cell count of 6,500/µL, and a platelet count of 420,000/µL. Other biochemical investigations, including hepatic and renal profiles, were within the normal range.\nIn view of the aforementioned clinical features, the patient underwent CT of the brain to rule out any space-occupying lesion. The CT demonstrated a homogenously hyperdense well-defined round lesion located in the midline at the approximate location of the foramen of Monro with prominent lateral ventricles (Figure ). This finding prompted a further investigation with MRI which redemonstrated the lesion as having an increased T1 and iso T2 signal intensity with the mass (Figure ). Such findings conferred the diagnosis of a colloid cyst.\nThe patient was prepared for an emergency neurosurgical operation for excision of the cyst. She had a right craniotomy with resection of the cyst using the transcallosal approach. The patient tolerated the procedure well without any complications. Histopathological examination of the resected cyst showed a ciliated pseudostratified epithelium lining of the cyst (Figure ). She had complete resolution of the headache and was discharged on the sixth postoperative day. |
An 11-year-old African American boy presented to the emergency department at our institution for an injury to his left knee which he sustained during basketball. He sustained the injury when trying to take off to shoot the basketball. Physical examination and radiographic evaluation confirmed a displaced tibial tuberosity fracture (). Based on Ogden classification, the fracture pattern was Type III B [].\nThe patient was taken to the operating room within 12 hours of his presentation, and open reduction and internal fixation of the tibial tuberosity fracture were performed using two 4.0 mm cannulated screws (). Postoperatively, an above-knee cast was applied for 4 weeks. Following cast removal, he started physical therapy for about 6 weeks for range of motion exercises and strengthening of his leg. The fracture healed uneventfully, and 6 months later, he was discharged to full activities without restrictions, with advice to follow up if needed.\nThree years later, he presented to our clinic with a progressive deformity and anterior knee pain, which was limiting his ability to play football, basketball, and skateboarding. Physical examination in standing and supine position revealed 30 degrees of hyperextension of the left knee compared to 5 degrees of hyperextension of the knee on the right side (). He had tenderness to palpation around the tibial tuberosity. Evaluation of the lateral radiograph revealed closure of the anterior aspect of proximal tibial physis and tibial tuberosity physis with hardware in situ. The proximal tibial geometry was altered with an anterior tibial slope of 13 degrees and the absence of the normal tibial tuberosity prominence (). Magnetic resonance imaging of the knee showed no evidence of chondral, ligamentous, or degenerative changes. The patient was recommended surgical intervention to correct his deformity.\nAt surgery, the tibial tuberosity and proximal tibial shaft were exposed using previous skin incision. The screws from the previous surgery were removed with assistance of fluoroscopy and debridement of the overlying bone. An extended, broad-based tibial tuberosity osteotomy was performed. The osteotomized fragment was retracted proximally. Two K-wires were inserted through the tibial tuberosity osteotomy site, directed from anterior distal to posterior proximal, starting about 3 cm below the joint line and directed obliquely to just superior to the proximal tibiofemoral joint (). The K-wires established the plane of the proximal tibial osteotomy. Using a combination of drill, oscillating saw, and osteotomes, a proximal tibial osteotomy was performed with a posterior based hinge. The osteotomy site was gradually opened to about 15 mm using tapered wedges as per the preoperative plan (). Bicortical iliac crest bone graft was harvested from ipsilateral ilium and was impacted into the opening wedge osteotomy site. A 4-hole, 15 mm trapezoidal-wedged tibial osteotomy plate (Arthrex, Naples, FL) was placed on the anteromedial aspect of the tibia and secured using two 6.5 mm proximal screws and two 4.5 mm distal screws. The osteotomized tibial tuberosity fragment with its attached patellar tendon was replaced over the proximal tibial opening wedge osteotomy site. To maintain the patellar height, the tibial tuberosity fragment was kept aligned at its proximal osteotomy cut, which moved its distal end proximally by about 15 mm. The fragment was tentatively stabilized using two 3.2 mm drill bits (). The position of the tibial tuberosity and tracking of the patella were confirmed. Definitive fixation of the tibial tuberosity fragment was then performed using a 6.5 mm cancellous screw proximal to the opening wedge osteotomy and a 4.5 mm cortical screw distal to it. A prophylactic fasciotomy was performed over the anterior compartment of the leg. Postoperatively, a knee immobilizer was used. The patient was advised nonweight bearing on his left lower extremity. Physical therapy was started 2 weeks after surgery for passive range of motion and static quadriceps exercises. His weight bearing status was advanced after the 6th postoperative week. At the latest followup, eighteen months after his second surgery, the patient had no complaints and was back to sports with no limitation of activities. Radiographic evaluation showed a posterior tibial slope of 4 degrees () which correlated with restoration of sagittal balance on clinical examination (). There was no significant change in the patellar height between preoperative measurements (Insall-Salvati ratio: 0.79, Blackburne-Peel ratio: 0.80) and measurements at final followup (Insall-Salvati ratio: 0.81, Blackburne-Peel ratio: 0.82). |
We describe the case of a 27-year-old white woman who had experienced an emergency caesarean delivery at 39 weeks for fetal distress with no postpartum complications. As part of our ongoing study “Vaginal delivery after caesarean section”, she underwent saline contrast sonohysterography 6 months after the caesarean section. The caesarean scar had a small indentation and the remaining myometrium over the defect was 7.5 mm (Fig. ).\nIn the current pregnancy, she had a dating scan at around 11 weeks with no remarks. She came for a transvaginal ultrasound examination at around 13 weeks as part of our study. This scan revealed a duplex pregnancy with one viable intrauterine fetus with normal anatomy and placenta located high on the anterior wall and a small gestational sac (8 mm) with a yolk sac without embryo was located in the caesarean scar (Fig. ). There was no extensive vascularity surrounding the sac. One corpus luteum was found in each of the two ovaries. She was asymptomatic.\nShe was informed that not enough evidence existed to advise a specific management of this condition. After discussion with her and her husband, expectant management was chosen with a new ultrasound examination after 5 weeks.\nShe came to our ultrasound department at 18 weeks, 22 weeks, and 30 weeks of gestation. She remained asymptomatic. The ectopic gestational sac was not visualized with transvaginal or transabdominal scans at the 18 weeks examination (Fig. ). The niche in the scar and the thickness of the thinnest part of the remaining myometrium appeared unchanged at all visits. The intrauterine pregnancy developed normally with no signs of abnormal placentation. At 30 weeks of gestation the ultrasound appearance of the scar area did not indicate any contraindications for vaginal delivery. The thickness of the lower uterine segment (LUS) was 4.9 mm (Fig. ). In agreement with our patient, vaginal delivery was planned. The staff of the labor ward was fully informed.\nShe was admitted to the labor ward with irregular contractions in week 37 + 0. Her cervix dilated to 3 cm with no further progress. Due to that oxytocin augmentation was administered for 3 hours. The duration of active labor was 6.5 hours. A healthy male neonate weighing 2985 g was delivered, with Apgar scores 9–10 at 1 and 5 minutes and umbilical cord pH 7.27. The placenta delivered spontaneously and total blood loss was 250 ml. The postpartum period was without any complications, and she was discharged home the next day.\nAt a follow-up visit 6 months postpartum, saline contrast sonohysterography showed no signs of the previous CSP, and the remaining myometrium over the hysterotomy scar defect was 5.7 mm (Fig. ).\nEthical approval for the ongoing study was obtained by the Ethics Committee of the Medical Faculty of Lund University, Sweden, reference number 2013/176. Our patient has given permission for publication of this case report in a scientific journal. |
A 9-year-old girl presented to our orthopaedic clinic, with main complaint of a mass which was getting bigger on her upper back. Her parents noticed that she has already had the mass since six years. The initial mass size was around 1 cm in diameter (like a marble) located just below the tip of both scapulae. Subsequently, more masses appeared on the left lateral side of vertebrae. It was accompanied by difficulty in moving both shoulders and neck since 4 years. Because of these complains, her family tried various traditional medicines without improvement.\nSix months prior to admission, she fell down on her back, and since then the masses grew progressively. She also felt pain around the masses. Meanwhile her neck and shoulder movement were becoming more limited than usual. She was brought to a district hospital in other province and then referred to our hospital to get more appropriate treatment. There was no history of similar disease in her family.\nOn physical examination, general condition was good and no abnormality was found in other organ. We found multiple bony prominences measuring about 2 cm in diameter at paravertebral region from cervical to lumbar and posterior thoracic wall. They made bone bridges starting from inferior occipital bone to cervical spine and extending to paravertebral lumbar region. We also found multiple masses at both axilla. Cervical lordosis and thoracic kyphosis were diminished. All the masses were hard in consistency. There was no tenderness, and the masses were fixed to the bone. The shoulder, cervical, and thoracal spine ranges of movement were limited ().\nRadiograph of the cervical spine showed new bone formation starting from occipital bone, cervical, until upper thoracal spine. Chest and shoulder radiographs also showed new bone formation at both humerus bridging to the tip of scapulae, with subluxation of both humeral head superiorly. The radiologist saw it as mustiple exostoses (osteochondroma lesion) with the differential diagnosis progressive myositis ossificans. HLA-B27 was negative, it was investigated since we also thought ankylosing spondylitis as a differential diagnosis. The 3D computerized tomography (CT) reconstruction revealed multifocal ossification on soft tissue including right pectoral muscle, bilateral latissimus dorsi, and left longisimus thoracis muscle. It was in accordance with myositis ossificans progresiva (). Magnetic resonance imaging (MRI) showed similar result with CT scan and no other abnormality detected at the spine.\nBecause of the problem was multiple exostoses and stiffness, total excision of occipito-cervico-lumbar and paravertebral ossification and also exostoses at bilateral shoulder was done. Histopathological findings showed tissue consisting of cartilaginous component, irregular underlying bony trabeculae, and bone marrow between trabeculae. There was also endochondral ossification with no cellular atypia (). After review of all patient’s data in one of the clinicopathological conference, we concluded that it was fibrodysplasia ossificans progressiva (FOP). Four months after surgery local recurrences were detected, which was confirmed with radiograph (). At three years follow up, she had similar signs and symptoms as before surgery. |
A 9-month old boy presented at a hospital in a south western state of Nigeria, with a swollen left upper arm adjoining the chest, low-grade continuous fever (38.1 °C), frequent passage of loose watery stool and persistent cries for more than 3 h. Child had been immunized about 24 h earlier. The mother reported that the symptoms were observed 2 h after the child was vaccinated with the measles vaccine at a private hospital. The child was one of three children reported to have been vaccinated with measles vaccine at a private hospital during the immunization clinic session.\nOn examination, he was mildly pale, febrile and anicteric. He was moderately dehydrated; mildly dyspnoeic with normal heart sound, heart rate of 148 beats/ min, breath sound was vesicular and respiratory rate of 54 cycles per minute. He was well nourished as the weight was appropriate for age. There was extensive swelling with skin discolouration (hyperemia) involving the entire left upper arm, sparing the distal third of the forearm and hand. There was also swelling of the upper part of the anterior chest wall. The swelling was firm and mildly tender. There was no history of adverse reaction to immunization or any form allergic reaction.\nA day after admitting the child, extensive erythema of the left upper arm and anterior area of the chest was observed with extensive scalded skin lesion involving the deltoid area, the upper chest wall and arm (Fig. ). Desquamation of the affected areas was observed presenting like severely burned skin from a hot liquid. There was darkening and hardening of the skin over the affected area on the arm with eventual severe necrosis up to a depth of about 5 mm thereafter (Figs. and ). A diagnosis of severe necrotizing fasciitis was made.\nRadical debridement of necrotic tissues was carried out under general anaesthesia. Child was also transfused with blood. Daily dressing of the wound was done and antibiotics administered were intravenous metronidazole (20 mg/ kg/ day in 3 divided doses) and ceftazidime (100 mg/ kg/ day in 3 divided doses). Child was referred to University College Hospital, Ibadan, a teaching hospital in a neighbouring state where skin grafting was performed. Presently, child have recovered and he is fully healthy.\nA causality assessment was conducted by the state AEFI committee using the detailed AEFI investigation forms using WHO AEFI causality assessment methodology [, ].Visits were made to the private hospital where the child was reported to have received the vaccine. The routine immunization focal person in the facility was interviewed. Assessment of available cold chain devices for vaccine storage was also carried out. The knowledge and skills of health workers in vaccine handling, management and administration were assessed [–]. In addition, the caregivers of two other children immunized during the session were recalled and interviewed. The case of interest was the first child to be vaccinated with measles vaccine during the immunization clinic while the second child, a 9 months old female who received vaccination from the same measles vial had fever and abscess formation at the site of immunization only however, the third child who was also vaccinated during the immunization clinic was healthy and without symptoms. The third child was found to be vaccinated with measles vaccine from a newly reconstituted measles vaccine vial different from the measles vaccine vial used for the other two children on the day of the immunization clinic. Incision and drainage procedure was carried out for the second child with wound dressing conducted for two weeks who thereafter recovered fully.\nThe findings from the investigation indicated that a programmatic error may have been responsible for the reactions.We found that two children were vaccinated with a measles vaccine that have been reconstituted for a period of > 6 h. The measles vaccine administered to these children was reconstituted 7 days ago and used during the previous immunization clinic with the left-over stored in a refrigerator within the hospital. This was due to poor knowledge and skill in vaccine management and administration among health workers who administered the vaccine. Other key issues identified includes poor documentation of vaccination activities using the recommended data management tools resulting in difficulty to tracked other children vaccinated with other vaccines for further investigation and poor vaccine storage system at the private hospital as the hospital lacks the recommended Solar Direct Drive (SDD) refrigerator for proper vaccine storage. Also, effort to retrieve the samples of the left-over doses of the vaccine in the opened vials for laboratory investigation proved abortive as the used/empty vial of the vaccine was said to have been discarded by the health workers immediately after the immunization clinic. Furthermore, blood samples collected from the child with NF by the attending physician during the preliminary case management at a local hospital for microbiological culture investigation shows contamination of culture plate as samples were not properly stored during the culture process due to lack of the required facility to perform the test at the hospital. |
34 years old patient, Gravida 6 Parity 3, previous 2 miscarriages (18 weeks & 12 weeks), was seen first at 23 weeks 4 days of pregnancy. She had undergone previous 3 cesarean sections and an evacuation of retained products of conception by curettage in 2013 for partial hydatidiform mole. At 27 weeks 5 days, she was admitted for vaginal bleeding. On further evaluation by ultrasound (), the diagnosis of placenta percreta was made (later confirmed by MRI). At 29 weeks, she had constipation with 2 episodes of urinary retention and she was put on continuous bladder drainage. She developed urinary tract infection and treated with appropriate antibiotics based on culture sensitivity. She continued to have repeated bouts of vaginal bleeding of varying amounts and severe constipation from 31 weeks of gestation.\nAt 32 weeks 4 days, patient underwent cystoscopy, which had shown signs of cystitis with no definite infiltration. She underwent classical cesarean section under combined anesthesia (Epidural + General). The umbilical cord was tied near insertion and the placenta was left in situ because there was no spontaneous separation. Then, the uterus was closed. Prophylactic temporary bilateral internal iliac artery balloons were inserted and inflated earlier. Uterine artery embolization was performed post cesarean section and selective angiograms confirmed adequate positioning. The patient required large volume of particles and still had incomplete embolization with the lower part of the uterus still showing some unblocked branches on both sides.\nPost-operatively, she was transferred to labor ward and within 4 hours, she developed clinical features of pulmonary embolism (PE). Some of her symptoms included drop in O2 saturation to 81%, tachycardia, chest pain, peripheral cyanosis, and signs of respiratory distress. Then, she was transferred to ICU and was initiated on heparin infusion. On chest X-ray, she had no atelectasis, pneumothorax, or pleural effusion. An immediate CT scan did not show any PE. There was no Doppler evidence of venous thrombosis in the femoral and popliteal venous systems. Later on day 1 post-operative, she had focal patchy consolidation left base and was started on parenteral meropenem, linezolid and fluconazole for the next 5 days. She had two consecutive CT scans on post-operative on days 2 and 3, which were negative. On ECG, there was right heart strain. She was now on enoxaparin. On the post-operative day 5, she was prescribed parenteral piperacillin-tazobactam for 5 days and she was shifted out of ICU next day. She had 500ml vaginal bleeding on the 9th post-operative day. 2 units PRBC were transfused. She was switched to oral cefuroxime and metronidazole and planned to continue on long-term low dose antibiotic. On post-operative day 11, she received methotrexate. On day 12, the MRA had shown the placenta was still enhancing with some areas of infarct and separation, fluid collection in the uterine cavity (present from day 1 post op, not increasing), with large ovarian veins, hugely distended and extensive pelvic varices, R>L, extensive collaterals. Her CRP was 12.7 mg/L.\nOn post-operative day 13, she underwent total abdominal hysterectomy. Intraoperatively, the bladder was densely adherent, drawn up, with large vessels in the broad ligament. The lower segment was bulging due to the presence of the placenta. The uterus was about 24 weeks’ size with adherent omentum. There was 100 mL of old blood in the cavity and the placenta was partially infarcted. The total blood loss was 2000 mL.\nPost-operatively, she was in ICU for 2 days receiving anticoagulation treatment (bridging treatment with enoxaparin + warfarin) and patient controlled analgesia. She had a bout of severe cough on day 4 and loose motions on day 5. She was diagnosed with vault hematoma, which was retro-vesical, about 120 ml in volume, treated conservatively. On day 10 she had been discharged from the hospital.\nShe presented to the ER on the post-operative day 16 and was diagnosed with chronic pulmonary embolism. Patient had a pulmonary embolus within the right middle lobe pulmonary artery; areas of sub-segmental embolus within the right lower lobe pulmonary arteries. She had no pleural effusions or consolidation and no mediastinal lymphadenopathy. She was readmitted for 4 days. She was started on therapeutic enoxaparin + warfarin. She was continued on 6 mg warfarin for 4 weeks after discharge. |
A 76 year old woman with a history of left flank bulge for 6 months presented with left-sided back pain in the same area for two months. She had no previous surgery or trauma in that area. On physical examination she had a 6 × 5 cm reducible bulge at the posterolateral part of left flank below the 12th rib. A CT scan showed herniation of retroperitoneal fat and descending colon through the fascial defect at the left superior lumbar triangle (). We planned the laparoscopic TEP approach for herniorrhaphy of this superior lumbar hernia, Grynfeltt hernia.\nUnder the general anesthesia the patient was placed in a full right lateral decubitus position with a lumbar roll in place. A 2 cm transverse incision was made over the hernia defect of the already reduced hernia which was below the 12th rib along the mid axillary line. The retroperitoneal fat was detached first with blunt dissection by an index finger and then with smooth instrument to create space in the retroperitoneal cavity. An 11 mm trocar was inserted and the space was expanded by a balloon dissector (Tyco Healthcare, Norwalk, CT, USA). Under the direct visualization with a 30° 10 mm scope, three 5 mm trocars were inserted anteriorly along the anterior axillary line approximately 5 cm apart (). Then, the 11 mm trocar was removed and the wound was plugged with gauze to maintain the pneumoretroperitoneum. With 30° 5 mm scope in the central port the retroperitoneal fat and structures were detached from the hernia defect and surrounding muscular-bony structures using 2 working ports above and below the central port (). A 15 × 15 cm octagonal polypropylene mesh () was inserted through the reinserted 11 mm trocar previously used for space making and placed on the lumbar wall to cover at least 4 cm margin around the hernia defect. The mesh was then fixed with transfascial sutures of polypropylene at the center and the margins of each quadrant. To secure the mesh in place we used Tacker (Tyco Healthcare). The mesh was not fixed to the iliac crest or the 12th rib. Postoperative pain was controlled with injections of analgesic (non-steroidal anti-inflammatory drugs) until the postoperative second day. There was no seroma or other wound complication. She was discharged on the 5th day after the operation (). There was no evidence of recurrence on follow up at 11 months. |
A 34-year-old south Indian female came to our outpatient department with complaints of insidious onset blurring of vision with gradual progression in the right eye for 2 months. The fall in vision was not associated with any pain or redness. She gave us a history of a similar episode of blurring in the same eye 3 years back, for which she was treated with intravenous methyl prednisolone followed by oral steroids following which her vision returned to normal.\nShe was diagnosed to have scleroderma a year back, following a skin biopsy from her forearm which showed features suggestive of scleroderma. At that time, she had hyperpigmentation over her ear lobules and nose and also suffered from gastritis and dysphagia. She was treated with pulse cyclophosphamide and then later shifted to oral mycophenolate mofetil (500 mg) twice a day by her rheumatologist. She was gradually withdrawn from systemic medications following the recovery of her systemic condition. She was not on any medications for more than 6 months when the current ocular symptoms started.\nOn ophthalmic examination, the best-corrected vision was 20/30 in the right eye and 20/20 in the left eye. Ocular motility was normal. Slit-lamp examination of the right eye showed quiet anterior chamber and anterior hyaloid face. Examination revealed normal sized pupil reacting well to light, and there was no relative afferent pupillary defect. The rest of the anterior segment examination was within normal limits. Fundus examination of the right eye showed a clear vitreous cavity with an edematous hyperemic optic disc along with multiple hypopigmented patches scattered throughout the posterior pole, suggestive of choroiditis []. The left eye anterior and posterior segment examination was normal.\nHer routine blood investigations showed a hemoglobin level of 10.6 g%, raised erythrocyte sedimentation rate of 38 mm/1 h. Peripheral blood examination showed hypochromic, microcytic red blood cells with occasional teardrop cells. Renal function tests which included blood urea and serum creatinine was done and the values were within normal limits. Test for antinuclear antibody was positive. Magnetic resonance imaging of the brain and orbit revealed no abnormality and a normal posterior coat of eyeball. She was also examined by our in house physician and rheumatologist, and all her systemic examination was within normal limits. Fundus fluorescein angiography (FFA) showed hypofluorescent areas in the early phase which corresponded to the hypopigmented lesions seen clinically, which increased in intensity in the late phase along with disc staining [].\nWe reached on a working diagnosis of right eye choroiditis with optic nerve involvement and started her on a course of oral steroids (1 mg/kg bodyweight) to which she responded dramatically. The steroids were gradually tapered over the next 3 months during which she did not have a relapse. This time we did not initiate her on any immunosuppressive drugs.\nAt her follow-up at 5 weeks, the right eye fundus lesions resolved and the vision improved to 20/20 []. The patient is on a regular follow-up with us and rheumatologist for the last 2 years, with a stable vision of 20/20 in both her eyes, with no episodes of recurrence. Her systemic condition is also stable and is currently sans all medications. |
A 34-year-old Caucasian female, who had originally undergone an uneventful laparoscopic adjustable gastric band 4 years ago, presented to the bariatric surgery clinic with inability to tolerate solids. A work-up revealed that the laparoscopic band remained in good position but the patient had oesophagitis and gastritis, causing swelling of the mucosa at the band site. The fluid was removed from the reservoir, and the patient was treated conservatively with anti-reflux medication and a full liquid diet. After 2 weeks of treatment the patient’s symptoms improved. After careful consideration, she wished to undergo revisional surgery converting the laparoscopic adjustable gastric band to a laparoscopic vertical sleeve gastrectomy. The patient moved through the appropriate multidisciplinary team approach and was found to be an appropriate candidate for surgery. She underwent laparoscopic removal of the adjustable gastric band and conversion to a laparoscopic vertical sleeve gastrectomy without complications. Her post-operative course was uncomplicated and she was discharged on post-operative day 3.\nOn post-operative day 12, the patient was readmitted to an outside tertiary care hospital for lightheadedness and shortness of breath and was found to have leukocytosis, with white blood cell count of 18,000 cells μl–1. The work-up included a CT scan with intravenous contrast of the chest, abdomen and pelvis, and the patient was diagnosed with a pulmonary embolism. The patient was immediately transferred to our centre for definitive care. When the patient arrived at our centre, the CT films from the outside hospital were reviewed by our radiologists and there was concern that there was air and a faint suggestion of oral contrast outside of the suture line (). Given this finding, an UGI evaluation was ordered. During the early phase, no leak was observed, owing, in part, to the slow passage of 30 ml oral non-ionic contrast (). Some residual contrast from the outside hospital CT was present in the transverse and descending colon. Only after delayed imaging and with administration of additional non-ionic contrast for a total of about 65 ml (approximately 2 h after the start of the fluoroscopic examination) was there a faint suggestion of extravasated contrast, best seen below the left hemidiaphragm (). Follow-up CT scan with oral contrast confirmed the obvious leak ().\nThe patient was treated definitively with endoscopic stent placement and clipping using an Ovesco clip (Ovesco Endoscopy AG, Tubingen, Germany) to close the leak. After an extended hospital course, she was discharged and is presently doing well. |
This is a 40-year-old female patient diagnosed with lower extremity CRPS type-I in the right ankle and foot, based on the IASP Budapest Criteria.[ The patient did not have any history of psychological disorder, seizure disorder or neurological abnormality. Physical examination in the first presentation showed the presence of well-healed incision scars from the previous surgeries in the dorsum of the right ankle and foot. There were mild color changes. There were moderate skin texture changes and trophic changes in the nails. The patient had excessive sweating both on inspection and palpation in the entire right foot. There was hyperesthesia and hyperalgesia of the entire dorsum of the foot (Fig. ). Right foot skin temperature was 3°C colder than the left foot. The patient had 4/5 weakness of the right foot extensors and toe flexors. Right ankle range of motion was moderately limited secondary to pain. The patient did not have any visible or reproducible involuntary motor movement in her history or physical examination. The patient underwent a first LSB with the same technique and medications as described in Case 1, but on the right side. LSB was performed under local anesthesia and the patient was given lorazepam 1.5 mg orally as a sedative prior to procedure. Temperature measurements of bilateral plantar skin as well as pulse amplitude of the right big toe with pulse oximetry were monitored continuously. Adequate sympathetic blockade was achieved after the block with confirmation of at least 2°C increase from the baseline temperature. The patient developed muscle spasms of the entire ipsilateral lower extremity in the recovery room 15 minutes after the completion of the first LSB (Video 2). These muscle spasms were not as intense as the Case-1, but significant enough that the patient was complaining of increased pain and was unable to stand. The patient was initially treated with 2 mg of midazolam IV. The patient did not respond to treatment with midazolam. As the presentation was very similar to Case-1, the patient was treated with IV DPH 50 mg. The patient responded immediately with abrupt resolution of muscle spasms. |
A 61 year-old Caucasian woman was referred to our colorectal clinic with an 18-year history of severe intermittent anal pain and constipation. She described experiencing intermittent anal spasms lasting around 15 minutes. These episodes were worse when sitting down for longer than 45 minutes or when lying in bed. The frequency of these anal spasms was increasing with time and occurring every hour at night at the time of presentation. Her constipation symptoms constituted experiencing difficulty in defecation and a sensation of incomplete evacuation. She had no response to amitriptyline or topical diltiazem. Her past medical history was unremarkable apart from four normal vaginal deliveries. Her sister had colorectal cancer diagnosed at the age of 49 and had previously been treated for an undiagnosed anal sphincter problem. There was no other relevant history of note.\nShe initially underwent a flexible sigmoidoscopy and magnetic resonance imaging (MRI) of her perineum. The endoscopy was reported as normal, whereas the MRI showed edema of the IAS. She subsequently had an endoanal ultrasound which confirmed that her IAS was abnormally thick and greater than 5 mm (Fig. ). Anal manometry revealed that although resting and squeeze pressures were within normal limits there were periods of a significant increase in anal resting pressure lasting longer than 2 minutes (Fig. ). Pressures during this period were in excess of 200 mmHg which settled spontaneously. These pressures were even higher than the maximum recorded squeeze pressure (Fig. ).\nShe had an examination of the anal canal under anesthetic which showed a very prominent sphincter complex. She also received Botox injections (Dysport™) at the 3 and 9 o’clock positions of the IAS which led to no subsequent resolution of her symptoms. She then underwent a lateral internal anal sphincterotomy by dividing half of the length (1 cm) of the IAS on the left lateral aspect. A biopsy of the IAS taken at the time of surgery was sent for histology which confirmed polyglucosan body myopathy of the IAS (Fig. ). At 3-month follow-up, she had complete resolution of her symptoms and has not contacted our department with any concerns for more than 1-year postoperatively. |
A 33 year old male patient presented with complaints of pain in the anteromedial aspect of the right knee. The pain had been ongoing for the last 4 years, but increased in severity for the last 8 months. The pain was exacerbated by movement of the knee joint, particularly when getting up from a chair and climbing or descending stairs. The patient described hypersensitivity in the infrapatellar region and was unable to wear tight fitting trousers, as even the slightest touch aggravated the pain. The patient was otherwise fit and well, with no history of trauma or surgery to the knee. Physical examination revealed a positive Tinel's sign on the medial aspect of the right knee, approximately 6 cm proximal to the joint line. Pressure over this area reproduced the patient's pain, which he described as “shooting” in nature.\nRadiographs of the knee joint revealed no bone abnormality. A magnetic resonance scan revealed early degenerative changes within the posterior horn of medial meniscus and a Grade I injury of the medial collateral ligament. The patient was started on a course of physiotherapy and analgesics, but these measures failed to relieve the pain. In view of the specific point tenderness on examination, we considered nerve entrapment or neuroma a possibility and, therefore, decided to explore this area.\nAt surgery, the infrapatellar branch of the saphenous nerve was found traversing the sartorius muscle at the site of maximum tenderness and making a tight angle on its way to the skin of the infrapatellar area [Figures and ]. We identified this as the likely cause of pain and partially divided the sartorius muscle in order to free the nerve, so that it lay in a straight line course without any tension or points of compression []. The patient described the relief of his pain immediately postoperatively and this has continued until his most recent followup at 6 months. Initially, he had mild numbness in the infrapatellar region, which has since recovered. |
A 71-year-old woman was referred to our hospital with a two-week history of cervical pain and numbness in the left upper limb. Neurological examination showed that her left upper limb had a slight motor weakness, with a manual muscle test score of 4. Hoffmann and Wartenberg reflexes were positive in both hands, and deep tendon reflexes in all extremities were hyperactive. However, the patient's left hand displayed reduced dexterity. MRI revealed cervical disc herniation, which compressed the left side of the spinal cord primarily at the C5-6 disc level (). Analysis of CSF obtained by a lumbar puncture showed no apparent abnormality and normal protein levels. CSF pressure was not measured. A diagnosis of cervical myelopathy due to cervical disc herniation was established, and an anterior cervical discectomy and fusion (ACDF) at C5-6 was performed.\nDuring the surgery, unexpected CSF leakage was observed when the posterior longitudinal ligament was incised with a scalpel. The dura mater was observed to be intact after the removal of the herniated disc. Thus, it was determined that the membrane that was incised when the CSF leakage occurred was not the dura mater. Fibrin glue was used on the surface of the dura, and no additional treatment of the CSF leakage was performed. Autologous iliac bone was grafted and fixed with a plate and screws. A postoperative X-ray image of the cervical spine revealed a black line posterior to the cervical vertebrae from the bottom of the C2 vertebra to the middle of the C5 vertebra (). Preoperative MRI and CT myelography images were carefully reviewed, and CSF collection on the ventral side of the dura expanding from the C2 vertebra to the T6 vertebra was identified (). CT myelography is routinely performed 30 minutes after injection of contrast medium. CT myelography was also performed after the surgery and showed that the CSF collection at the surgical site disappeared. However, the CSF collection persisted in the region between levels T2 and T6. An MRI performed 6 months after the surgery showed continued CSF collection between levels C7 and T6. However, no spinal cord compression was observed. Neurological symptoms were relieved after the surgery, and no symptoms related to the CSF leakage were observed. Although the patient was asymptomatic at one year postsurgery, an MRI of the brain was performed, which did not show signs of siderosis, brain sagging, meningeal thickening, or any other abnormality. |
A 32-year-old man reported to the department with the chief complaint of pain and swelling below the left eye obstructing the vision from the same eye from past 1 month and an ulcer on the roof of mouth from the past 15 days. Pain was gradual in onset, severe, throbbing, continuous, and radiating to the left temporal region associated with swelling over the left maxillary sinus region that was insidious in onset and gradually progressed to the present size causing nasal obstruction and oedema around the left eye. There was a history of recurrent nasal discharge from the left nostril since 20 days. Around 15 days back, an ulcer developed over the palate which was rapidly increasing in size and was interfering with speech.\nPatient's medical history revealed that he was uncontrolled type 1 diabetic patient from past 2 years and was on treatment (Inj. Mixtard 14 units per day). There was a family history of diabetes as patient's father had type 1 diabetes. The patient was tobacco chewer from past 15 years.\nOn general physical examination, the patient appeared febrile and the BP was recorded to be 160/90 mmHg.\nOn extraoral examination, a difuse swelling was appreciated over the left maxillary sinus region extending superoinferiorly from the left supraorbital margin to a line joining the left corner of mouth to the left ear and anteroposteriorly from nasal bridge to left ear. Overlying skin appeared normal with no secondary changes and on palpation was soft to firm in consistency with diffuse borders and tender with no local rise in temperature [].\nOn intraoral examination, an infiltrating ulcer approximately 3 × 4 cm2 with irregular borders was appreciated over the hard palate. Ulcer was covered over by the necrotic slough and on the anterior aspect of the ulcer, part of the underlying bone was also exposed. Ulcer was nontender with few areas of erythema over the margins [].\nConsidering the patient's medical history and a rapidly proliferating ulcer denuding the underlying bone, a provisional diagnosis of deep mycotic infection of palate was made. The differential diagnosis of midline lethal granuloma and malignant ulcer was thought.\nLater the patient was subjected to blood investigations which revealed raised ESR (34 mm/Ist hour) and random blood sugar around 220 mg%. Tridot and HBs test were negative.\nWater's view revealed diffused radiopacity over the left maxillary antrum extending into left nasal cavity [].\nCT scan in both axial [Figures 4 and ] and coronal sections [Figures 5 and ] revealed radiopacities along the walls of the left maxillary antrum with focal destruction of the bony walls extending into the left nasal cavity and floor of the left orbit.\nThe histopathological report suggests the presence of numerous aseptate fungal hyphae branching at 90° with neutrophillic infiltrate invading the smaller blood vessels (PAS staining) [].\nBased on the above findings, final diagnosis of “Rhinomaxillary mucormycosis—A subdivision of rhinocerebral mucormycosis” was made.\nThe following treatments were carried out:\nSurgical debridement of the necrotic tissue. Amphotericin B 1 mg/kg/day for 1 month. Nebulisation |
A 62-year-old man presented at the Neurology consultation with a six-week history of a severe, strictly left orbitotemporal headache, with a frequency of three attacks per week, occasionally more than one at the same day. Most of them occurred in the first half of the night, waking him up, and lasted between thirty minutes and one hour. He used to take ibuprofen as acute treatment, with unsatisfactory response, since he did not notice a significant difference between treated and untreated attacks in terms of duration and pain intensity. To relief the pain, he used to open the window to get some fresh air. The headache was always associated with ipsilateral conjunctival injection and lacrimation. Pain triggers were not identified by the patient. He had no personal or familial history of headaches. His medical history was remarkable for hypertension and asthma, with a past surgical history including septoplasty and bilateral middle turbinectomy and uncinectomy due to nasal respiratory insufficiency. By the time of medical evaluation he was asymptomatic and neurological exploration was unremarkable. The clinical picture was suggestive of a CH and the patient was medicated with verapamil 120 mg daily. A MRI scan was performed, which revealed a sphenoid sinus mucocele, without secure expansion of the sinus. Two weeks later the patient came to the Emergency Department with complaints of horizontal diplopia that he noted when he woke up in that morning. He maintained the headache attacks, with similar characteristics, despite prophylactic therapy. Neurological examination revealed left eye adduction palsy and ptosis. A brain CT scan was performed and excluded lesions other than the mucocele. Paranasal sinus MRI revealed molding of the medial wall of left cavernous sinus by the sphenoid mass (). A paranasal sinus CT scan was also performed to allow for a better characterization of the lesion, showing sclerosis and interruption of the roof and posterior wall of the left sphenoid hemisinus (). The patient was submitted to surgical drainage of the mucocele by transnasal-transphenoidal approach, with complete resolution of the adduction impairment, persisting a mild left eye ptosis. After the surgery the attacks stopped, and in the six-month follow-up he reported no further attacks. |
This is a 30-year-old man who sustained an open, segmental right femoral shaft fracture and an ipsilateral femoral neck fracture after being hit by a vehicle while riding a scooter. He initially underwent irrigation and débridement of the open fracture, retrograde intramedullary nail fixation of the right femur, and dynamic hip screw fixation of the ipsilateral femoral neck (Figure ). The patient was instructed to be nonweight bearing on the right leg postoperatively; however, 3 weeks later, he sustained a fall, which resulted in failure of the intramedullary nail at the level of distal extent of the dynamic hip screw construct. The patient then underwent revision of the intramedullary fixation and stage 1 of the induced membrane technique after the segment of devitalized diaphysis was removed. The segmental defect of the femur diaphysis measured 9.3 cm medially and 4.8 cm laterally. During this period, the patient remained nonweight bearing on the right leg. One month after stage 1, the patient underwent stage 2 with the removal of the antibiotic spacer and placement of autograft bone from the iliac crest and proximal tibia. The patient was made nonweight bearing on the right leg for 12 weeks, advanced to toe-touch weight bearing for three more weeks, and finally progressive weight bearing. However, the patient continued to have pain at the fracture site. Nine months later, he was diagnosed with a right femur nonunion. He was referred to the author and underwent a repeat autograft bone grafting of the right femur defect with RIA (Depuy Synthes). At that time, placement of a fibular strut allograft was performed as described above. The patient was made toe-touch weight bearing after surgery and advanced to partial weight bearing of 25% to 50% body weight 6 weeks after. Ten weeks after surgery, he was advanced to partial weight bearing of 50% to 75% body weight. The patient was finally advanced to full weight bearing 5 months after the last surgery. At the last visit, the patient was fully weight bearing without pain and had knee range of motion from 0° to 100° (Figure ). The last clinical follow-up was obtained at 15 months postoperatively. |
An 85-year-old Caucasian male was admitted after sudden onset of expressive aphasia and weakness in both legs lasting 20 seconds. He was athletic, self-reliant and had no cognitive impairment. During the last 28 years, he had experienced 8-10 heterogeneous episodes of acute neurological symptoms, such as central facial palsy, hemiparesis, and non-fluent aphasia, lasting from seconds to 3-4 hours. Precerebral duplex and electrocardiography (ECG) were performed several times with normal results, and EEG registration and 24-hour Holter monitoring had been normal. Previous MRI scans showed no abnormal restricted diffusion, as seen in acute cerebral infarcts, but infarct sequelae in the left temporal lobe and both thalami. Several years later, three additional infarct sequelae were detected in the cerebellum. The patient was treated with platelet inhibitors, and medications and dosages were adjusted after new episodes. There was no suspicion of lack of compliance. Except from age, migraine, and previous smoking, with cessation 35 years ago, he had no known risk factors for cerebrovascular disease. On the current admission, he presented with reduced motor speed in his left arm and leg. Electrocardiography and Holter monitoring showed no signs of atrial fibrillation. CT and MRI revealed multiple, cortical infarct sequelae in the anterior and posterior circulation territories of both hemispheres, and MRI also detected two acute embolic infarcts in the right occipital lobe and one in the left parietal lobe (Figure ). CT and MRI angiograms and duplex sonography did not show significant plaques or stenoses, and pre- and intracerebral flow were normal with asymmetrical vertebral arteries, which were considered a normal anatomical variant. Cortical infarcts in several vascular territories strongly suggest cardioembolic etiology, but transthoracic echocardiogram showed no cardiac sources of emboli, and there was no sign of left atrial enlargement, which may be seen in the presence of atrial fibrillation. The patient concurred to further diagnostic tests aiming to determine the cause of recurrent cerebral emboli, although he was informed that the results would not necessarily alter treatment recommendations. We performed a transcranial Doppler (TCD) bubble test with 10 mL air-mixed saline injected into the left cubital vein while the left middle cerebral artery was insonated with a 2-MHz probe. Injection at resting state produced no microembolic signals, while injection after Valsalva maneuver resulted in a shower of microembolic signals followed by single signals persisting for over 30 seconds. The result implied the presence of a latent right-to-left shunt, and transesophageal echocardiography verified a large patent foramen ovale (PFO; Figure ). In agreement with the patient, we decided on non-operative treatment. Due to previous failure of antiplatelet treatment, we changed to a direct oral anticoagulant (dabigatran 110 mg twice daily), intended as a lifelong treatment. He had no subjective complaints at discharge. |
A 14-year-old female patient reported to our department with complaint of swelling in the gums of the upper and lower right hand side quadrants of the mouth since a year. The patient first noticed the swelling 4 years before in the upper right side of the mouth - gradually and slowly increasing swelling.\nThe patient had delayed tooth eruption of the upper and lower right premolar teeth associated with gingival swelling. She underwent excisional surgery for the same 2 years before at a private dental clinic. Postsurgery within 6 months, the patient again noticed the swelling which gradually increased in size. The patient gave history of rapid increase in size since the preceding 6 months and spread toward the teeth of the left hand side of the mouth. Swelling was painless, but the patient complained that it interfered with chewing.\nThere was no history of epilepsy or major illness. She was undergoing treatment for anemia with iron supplement. Developmental milestones and other systems of the child were normal. Family and menstrual history was noncontributary. The patient has unilateral mastication habit with left side since childhood. The right hand used for brushing.\nGeneral physical evaluation was done. The patient had normal physical appearance and psychomotor skills. Normal bone development seen on the chest and extremity radiographs. The patient showed slight facial asymmetry with fullness of the right upper lip. The lips were competent [].\nGingival enlargement was more predominant on the right hand side of the mouth, but mild involvement was present extending to the incisors as well as the lingual aspect of the left mandibular molar region [Figures and ]. Only the maxillary left posterior teeth were completely uninvolved. Both facial and palatal/lingual aspects were involved in the maxillary and mandibular right hand side quadrants covering almost the entire clinical crown. There was diffuse involvement of marginal, papillary, and attached gingiva. The swelling was irregular, largely pale pink, and firm devoid of stippling along with softened reddish pink areas toward the occlusal surface associated with calculus deposits.\nBleeding on probing was present with 14–16 regions. Several teeth were clinically submerged including the maxillary canines (13, 23) and mandibular left canine (33) and both the mandibular second premolars (35, 45). Even, the maxillary and mandibular second molar of the right hand side (17, 47) were clinically submerged. Deep pseudopockets along with attachment loss of up to 13–15 mm were present with 14, 15, 16, and 46 which showed Grade II mobility. Grade I mobility was present with 11, 12, 21, 22, 31, 32, 41–44.\nOrthopantomogram (OPG) and three dimensional cone beam computed tomography evaluation was done. The radiographs revealed that the submerged teeth had bony impaction except for the maxillary and mandibular right second molars which appeared to be still erupting. Severe bone loss was present with 14, 15, and 16 teeth []. In this region, there was evidence of increased spacing and further bone loss compared to an OPG taken 3 years prior and the current radiograph. In addition, generalized crestal bone loss was present except with the left maxillary posterior teeth.\nBlood investigations revealed the normal blood profile and hormonal levels. Hemoglobin level was low 10.3 mg%.\nAfter obtaining written consent, thorough scaling, root planing, and curettage were done and anti-microbial rinse chlorhexidine gluconate 0.2% prescribed and in addition used for irrigation of the pockets. This regime was repeated weekly for 3 weeks as it was difficult for the patient to maintain adequate plaque control. An incisional biopsy was done and 8 mm × 10 mm tissue specimen was sent for histopathological evaluation.\nPostphase I appearance of the tissues showed overall superficial reduction in inflammation. Following this phase, electrosurgical gingivectomy was performed. First, the maxillary surgery was performed. After administering nerve block anesthesia to the patient, the area of tissue to be excised was demarcated with a probe. The electrosurgical unit (megasurg - high frequency radiosurgery unit) was set up and the passive electrode – the patient plate tied to the patient's calf. The needle electrode (active electrode) at 8 MHz was used to outline the external bevel incision. The tissues were further undermined with a bar electrode and removed in bulk. The bar electrode was then used to plane the tissues and achieve a favorable tissue contour. Following excision, thorough irrigation of the site was done. In some sites, additional curettage was done along and closure of the area with sutures. The operative procedures for Maxilla are shown in . Periodontal dressing was placed, and the patient was prescribed anti-biotic and analgesic medication.\nThe mandibular surgery was performed 2 weeks later similarly []. Postoperative healing a week later was satisfactory. The case was followed-up for 8 weeks postoperatively, and then every 3 months for 2 years. Favorable healing of the sites were seen postoperatively at 1 week, 1 month, and 3 months intervals. The mobility of teeth had significantly decreased from Grade II mobility to physiologic by 3 months postoperatively. The patient was able to maintain good oral hygiene. The patient has visited for recall follow-up since the past 2 years with minimal evidence of recurrence [].\nEvaluation was performed using routine H and E staining as well as polarized microscopy with specific connective tissue staining to verify the nature of the fibrosis. Abundant collagen with hyalinization of the collagen fibers was seen. Congested blood vessels with mixed inflammatory infiltrate focally transgressing the overlying epithelium were observed. There was no evidence of dysplasia or malignancy []. |
A 57-year-old male presented with a 2-year history of swelling in the left leg and heaviness after walking. The patient had no history of comorbidities, trauma, or surgery, except alcoholic liver cirrhosis (Child class B). Physical examination revealed swelling of the left leg without tenderness. Lower extremity computed tomography venography (CTV) had been performed at another hospital for DVT and possible extrinsic compression and revealed multiple cystic lesions compressing the distal portion of the left SFV without DVT (). The proximal cyst had entwined the SFV from the posterior aspect to the medial and anterior aspects of the vein, and the distal cyst was positioned at the posterior aspect of the vein extending down to the level of the adductor hiatus. The cysts were not interconnected. An operation was performed considering the possibility of venous ACD. The SFV was exposed by a medial approach. Two cystic masses were detected along the distal SFV, and no joint connections were identified between the two cystic masses after a thorough investigation. The larger proximal cyst measured 1 cm×3 cm and the smaller cyst measured 1 cm×1.5 cm (). The cyst contained mucinous fluid without communication with the vessel lumen. The cyst was excised by resecting its roof portion first and then peeling off the remnant cyst in contact with the vein wall by curettage. During cyst excision, no transmural involvement was observed. After cyst excision, the prevailing stenosis of the SFV was observed, although the obstructive effect was relieved (). Postoperative histology revealed a cystic structure consisting of collagen fibers without any synovial cells; therefore, synovial cyst was excluded from the diagnosis (). Specific staining for synovial markers was not performed because of the absence of synovial cells. Despite the use of low molecular weight heparin (LMWH) (enoxaparin, 40 mg twice daily), leg swelling recurred 1 week after surgery. CTV revealed popliteal vein thrombosis beginning at the center of the knee joint space and extending up to the adductor hiatus (). After 3 months of oral anticoagulant therapy with rivaroxaban (15 mg twice daily for 3 weeks and then 20 mg daily), a follow-up CT scan revealed resolution of DVT with mild stenosis of the SFV and no recurrence of ACD (). The anticoagulant therapy was discontinued. Follow-up was performed every 6 months, and CTV was performed annually. During 3 years of follow-up, the patient did not present any symptoms such as leg swelling, redness, or cramping and had no recurrence of ACD and new DVT on follow-up imaging. |
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